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A factor analysis of tic symptoms in Gilles de la Tourette's syndrome.

OBJECTIVE: Gilles de la Tourette's syndrome is a well-characterized disorder with clear DSM-IV criteria. However, the great deal of clinical variability across patients may represent an underlying etiologic complexity. Issues of phenotypic heterogeneity are particularly critical to current efforts at mapping genes involved in this syndrome. METHOD: Lifetime tic symptom data were obtained from direct structured interviews of 85 Tourette's disorder probands. Information on 29 tic symptoms was elicited. The probands' tic symptoms were grouped by using agglomerative hierarchical clustering, with no a priori assumptions concerning the relatedness of symptoms. Scores for the probands' symptom clusters were used as variables in a principal-component factor analysis. The relationships of the resulting factor scores to comorbidity in probands and recurrence risks in relatives were examined. In addition, intraclass correlations were calculated for within-family factor scores of 36 families. RESULTS: Four significant factors were identified: 1) aggressive phenomena (e.g., kicking, temper fits, argumentativeness), 2) purely motor and phonic tic symptoms, 3) compulsive phenomena (e.g., touching of others or objects, repetitive speech, throat clearing), and 4) tapping and absence of grunting. Analysis of the symptom loadings, comorbidity, recurrence risks, and within-family correlations indicate that these factors represent a valid structure with clinical and biological relevance. CONCLUSIONS: In this symptom-based factor analysis of Tourette's disorder, four factors accounted for 61% of the phenotypic symptom variance in Tourette's disorder probands and their first-degree relatives. Three of these factors may indicate the presence of heritable components of the Tourette's disorder phenotype.

Adult↗

An investigation of tic suppression and the rebound effect in Tourette's disorder.

Many patients, parents of children with Tourette's disorder, and professionals have suggested that following a period of suppression, tics will rebound to a rate that will exceed the average rate of occurrence. At present, there are no empirical data to support or refute such an effect. This experiment utilized an A-B-A design with replication to test this hypothesized effect. Following baseline observation, participants were instructed to refrain from exhibiting tics while watching videotapes, engaging in conversation, or while alone in a room with no activity. Observation continued following the suppression phase. Results of this experiment showed suppression of tics in almost one half of all sessions, with adults demonstrating suppression more frequently. Furthermore, results of this experiment failed to support a commonly held perception that following a period of voluntary suppression tics will rebound to a rate that will exceed the average rate of occurrence.

Adult↗

Randomized trial of parent management training in children with tic disorders and disruptive behavior.

Oppositional, defiant, and disruptive behaviors are common in clinical samples of children with tic disorders. In this study, we sought to evaluate the short-term efficacy of a structured parent training program in children with tic disorders accompanied by disruptive behavior. Children with tic disorders and at least a moderate level of disruptive behavior were randomly assigned to a 10-session structured parent management training program or to continue treatment as usual. Twenty-four children (18 boys and 6 girls) between the ages of 6 and 12 years (mean 8.9 +/- 2.0 years) were enrolled; 23 subjects completed the study. At baseline, subjects showed moderate to severe levels of oppositional and defiant behavior. Twenty subjects (83%) were on stable medication. The parent-rated Disruptive Behavior Rating Scale score decreased by 51% in the parent management training group compared with a decrease of 19% in the treatment as usual group (P < .05). On the Improvement scale of the Clinical Global Impression, a rater masked to treatment assignment classified 7 of 11 subjects who completed parent management training as much improved or very much improved compared with 2 of 12 subjects in the treatment as usual group (Fisher exact test, P < .05). These results suggest that parent management training is helpful for short-term improvement in disruptive behavior problems in children with tic disorders. Larger randomized clinical trials are needed.

Analysis of Variance↗

Familial tic disorder, parkinsonism, motor neuron disease, and acanthocytosis: a new syndrome.

We report two brothers who were of consanguineous parents and who displayed a unique association of motor and vocal tics, parkinsonism, distal muscular atrophy, and acanthocytosis. In the older brother, leg weakness and muscle wasting started at age 13, and he became wheelchair bound at 40. Electrophysiologic studies and muscle biopsy confirmed diffuse denervation. Involuntary vocalizations and facial tics began at age 36, but within 5 years the tics were replaced by progressive parkinsonism with supranuclear ophthalmoparesis. CSF studies implied impaired central dopamine and serotonin turnover. In the younger brother, orofacial tics started at age 36, vocalizations and fasciculations in the legs began 1 year later, and parkinsonian findings were present at age 40. This is the first report of an association of Tourettism, parkinsonism, motor neuron disease, and acanthocytosis occurring as an autosomal recessive syndrome.

Acanthocytes↗

Gilles de la Tourette's syndrome: tics and central nervous system stimulants in twins and nontwins.

Thirty-four of 170 surveyed individuals with Tourette's syndrome (TS) were treated with CNS stimulants before age 18. In 24% of treated individuals, persistent exacerbation of tics was closely associated with treatment. In 3%, tic response was transient, and in 24%, tics were not obviously associated with treatment. Six pairs of monozygotic twins were discordant for stimulant treatment, and all untreated co-twins also developed TS. The number of individuals in whom stimulants permanently exacerbate tics may be small, but the risk appears to be real. Genetic vulnerability and duration and timing of treatment may mediate response.

Antipsychotic Agents↗

Sensory tics in Tourette's syndrome.

Sensory tics are localized uncomfortable sensations for which patients attempt to obtain relief by producing movements or vocalizations. We report 3 patients with Tourette's syndrome (TS) and sensory tics to illustrate this poorly recognized symptom. A survey of 34 randomly selected TS patients indicates that sensory tics are common and should be considered part of a clinical spectrum of tics and associated sensory phenomena.

Adult↗

Extracorporeal 'phantom' tics in Tourette's syndrome.

Some tics are intentional movements made in an attempt to diminish uncomfortable sensations. These sensations, sometimes termed sensory tics, are focal and usually arise in the part of the body involved in the subsequent motor act. We report a patient whose sensations associated with tics were felt in other people and in objects. The discomfort associated with his out-of-body sensations was temporarily relieved by touching or scratching the object involved. The definition of premonitory sensations should be expanded to include extracorporeal sensations, and a history of external sensations should be sought in patients with Tourette's syndrome. Theories on the generation of tics must incorporate an explanation for extracorporeal sensations.

Adult↗

Use of an antecedent analysis and a force sensitive platform to compare stereotyped movements and motor tics.

Stereotyped movements displayed by 6 participants and tics displayed by 6 children were evaluated using an antecedent behavioral analysis and a force sensitive platform. We found that tics occurred more often in an alone condition when compared to high preference toy and play conditions, whereas stereotyped movements were more variable across conditions. Using temporal measures and spectral analysis data from the force sensitive platform, we found that stereotyped movements tended to be longer in duration and displayed more rhythmic qualities when compared to tics. Results indicate that stereotyped movements and tics differ both quantitatively and qualitatively and support the use of new technologies, such as force plate technology, to more accurately evaluate these differences.

Antipsychotic Agents↗

Genetic and environmental influences on stuttering and tics in Japanese twin children.

The purpose of this study was to clarify the genetic contribution to stuttering and tics in childhood using the largest databases of Japanese twins. The subjects were 1896 pairs of twin children consisting of 1849 males and 1943 females with a mean age of 11.6 years (3 years to 15 years). All data were gathered by questionnaire. The prevalence of stuttering was 6.7% in males and 3.6% in females (p < .0001). The prevalence of tics was 6.8% in males and 4.1% in females (p = .0021). Concordance rates and polychoric correlations were all higher in monozygotic pairs than in dizygotic pairs irrespective of sex combination. Structural equation modeling showed that the proportion of total phenotypic variance attributable to genetic influences was 80% in males and 85% in females for stuttering, and 28% in males and 29% in females for tics. Moreover, co-occurrence between stuttering and tics was observed in 0.8% of males (tetrachoric correlation: r = .18) and 0.5% of females (r = .31), which was attributed partly (nearly 10% of total genetic variance of each trait) to the common genetic factors, with genetic correlation of r = .32.

Adolescent↗

Childhood caffeine tic syndrome.

OBJECTIVE: To determine whether caffeine ingestion was temporally correlated with tics in 2 healthy children. METHODS: Two first-degree cousins were observed over a period of approximately 3 years, and the presence and absence of tics was recorded and correlated with consumption of or abstinence from caffeinated foods or beverages. RESULTS: Appearance and disappearance of tics were closely and clearly temporally correlated with ingestion and elimination of caffeine in the cousins' diets. CONCLUSIONS: Our observations suggest that caffeine may precipitate tics in susceptible children.

Beverages↗

Tics in childhood.

The tic disorders represent a spectrum of disability ranging from simple transient tics of childhood to severe Tourette syndrome. Diagnosis depends on an awareness of their existence, willingness to accept the child and parents' histories, and appreciation of the broad range of symptoms. Although the etiology is not clear, some cases are familial. Environmental agents, especially psychostimulant drugs, may precipitate occasional cases. Treatment with haloperidol is useful for 80% to 85% of patients but associated with troublesome side effects in one-half of those treated. The long-term prognosis for complex tic disorders is not as bad as once thought, with improvement or remission often occurring in late adolescence. Abnormalities of central nervous system dopamine metabolism may be involved in the pathophysiology of the tic disorders.

Child↗

Delta 9-tetrahydrocannabinol (THC) is effective in the treatment of tics in Tourette syndrome: a 6-week randomized trial.

BACKGROUND: Preliminary studies suggested that delta-9-tetrahydrocannabinol (THC), the major psychoactive ingredient of Cannabis sativa L., might be effective in the treatment of Tourette syndrome (TS). This study was performed to investigate for the first time under controlled conditions, over a longer-term treatment period, whether THC is effective and safe in reducing tics in TS. METHOD: In this randomized, double-blind, placebo-controlled study, 24 patients with TS, according to DSM-III-R criteria, were treated over a 6-week period with up to 10 mg/day of THC. Tics were rated at 6 visits (visit 1, baseline; visits 2-4, during treatment period; visits 5-6, after withdrawal of medication) using the Tourette Syndrome Clinical Global Impressions scale (TS-CGI), the Shapiro Tourette-Syndrome Severity Scale (STSSS), the Yale Global Tic Severity Scale (YGTSS), the self-rated Tourette Syndrome Symptom List (TSSL), and a videotape-based rating scale. RESULTS: Seven patients dropped out of the study or had to be excluded, but only 1 due to side effects. Using the TS-CGI, STSSS, YGTSS, and video rating scale, we found a significant difference (p <.05) or a trend toward a significant difference (p <.10) between THC and placebo groups at visits 2, 3, and/or 4. Using the TSSL at 10 treatment days (between days 16 and 41) there was a significant difference (p <.05) between both groups. ANOVA as well demonstrated a significant difference (p =.037). No serious adverse effects occurred. CONCLUSION: Our results provide more evidence that THC is effective and safe in the treatment of tics. It, therefore, can be hypothesized that the central cannabinoid receptor system might play a role in TS pathology.

Adolescent↗

Prevalence of tics and Tourette syndrome in an inpatient adult psychiatry setting.

OBJECTIVE: Given the widely recognized genetic basis for Gilles de la Tourette syndrome (TS) and the suggestion that the putative TS gene(s) may be expressed as or associated with a variety of psychiatric illnesses, this study was undertaken to assess the prevalence of tics and TS in a psychiatric inpatient population. DESIGN: Cross-sectional study. SETTING AND PATIENTS: 200 consecutive adult patients who were admitted to the psychiatric wards of University College London Teaching Hospitals. OUTCOME MEASURES: TS and related behaviours, as assessed by the comprehensive semi-structured National Hospital Interview Schedule. RESULTS: None of the 200 patients had definite TS, but 2 were observed to have motor tics; 10 had a history of tics (present for less than a year), and 7 reported a family history of tics. Thus, 19 (9.5%) inpatients qualified for inclusion in a broadly defined TS diathesis. These rates are significantly lower than those reported in a similar community based epidemiological study of adolescents (p = 0.018). CONCLUSIONS: Our findings do not support the theory that TS and related behaviours are over-represented among adult inpatients with psychiatric illnesses.

Adolescent↗

[Paroxysmal stereotypy-tic-dystonia syndrome].

CASE REPORTS: We report the cases of four males from four different families, who presented paroxysmal episodes from the 1st 2nd year. These episodes were characterised by asymmetrical bilateral dystonia of the upper limbs, predominantly in both hands, and were associated with orofacial dyskinesias, stereotipies (jumping, arm flapping, etc.), facial tics and, occasionally, phonic tics. Consciousness is not affected in any of the cases. These movements are triggered in situations where the patient is relaxed or excited. They occur daily and last from a few seconds to 30 minutes. Between the bouts, they remain asymptomatic. Family cases suggest it is inherited by autosomal dominant transmission, perhaps linked to the X chromosome; in addition, two cases are sporadic. In the only adult, the movements progress to a series of rhythmic bilateral dystonic myoclonias and facial tics dyskinesias. All the studies carried out, EEG, hemogram, biochemical analysis, neuroimaging, copper and ceruloplasmin levels, were normal. CONCLUSIONS: 1. We report a non epileptic paroxysmal disorder originating in the extrapyramidal tracts with its own characteristics, with onset during early childhood, which is associated with stereotipies, tics and dystonia; 2. It occurs predominantly in males; 3. It is inherited by autosomal dominant transmission, or perhaps sex linked autosomal dominant inheritance, and there are also sporadic cases; 4. The range of clinical features is very wide and includes cases in which there are few symptoms to others where the extent and gravity of the disorder is very significant.

Adult↗

Tourette syndrome: not just a tic disorder.

Although tics are considered the hallmark of Tourette syndrome, arguably tics may not be the only or primary presenting symptom. For many children diagnosed with Tourette syndrome irritability, frustration intolerance, hyperactivity, inattention, ritual behavior or other difficulties may have been present a number of years before the appearance of tics. Children with Tourette syndrome are often highly co-morbid with attention deficit-hyperactivity disorder, obsessive compulsive symptoms, and other related behavioral problems that should be detected and treated effectively. Therefore tics should not be the sole indicator or receive over emphasis in the detection and treatment of Tourette syndrome.

Child↗

Pollakiuria in children with tic disorders.

BACKGROUND: To describe the rarely reported characteristics of pollakiuria in children with tic disorders (TD). METHODS: During a period of one year, all patients visiting our Tourette Syndrome (TS) Clinic with TD were studied for presentation of pollakiuria. RESULTS: We found that nine (1.9%) out of 467 children with TD had pollakiuria: 407 boys and 60 girls, with a mean age of 8.7 years. Of these nine children, there were eight boys and one girl, and their mean age was 7.4 years. Five had transient TD, one had chronic motor TD and the remaining three had TS. Their pollakiuria recovered within two to six months, except for two male patients aged six and 19 years, whose pollakiuria persisted for more than a year. Factors precipitating anxiety were identified in only three children. Obsessive compulsive behaviors could be traced in eight patients. CONCLUSIONS: Pollakiuria occurring in children with TD is not frequently encountered. It usually has a benign course, as it does in those without tics. However, sometimes the pollakiuria waxed and waned over years, as do tics. Pollakiuria could be a manifestation of tics or a compulsion, instead of just an anxiety disorder.

Adolescent↗

[Neuropsychological performance in ADHD and tic-disorders: a prospective 1-year follow-up].

Inhibitory deficits in the brain play a major role in the pathophysiology of ADHD and tic disorders. Impairment of executive functions (EF) may be directly linked to these abnormalities. Hence, parallel to an amelioration of symptomatology in ADHD and in tic disorders a decline of EF impairment should be observed. This phenomenon would indirectly suggest that there exist inhibitory deficits in these disorders. Age and IQ matched samples of boys one with ADHD (n = 15; age: M = 128 months SD = 16, IQ: M = 101 SD = 11), the other with tic disorders (n = 14; age: M = 132 months SD = 16; IQ: M = 101 SD = 10) have been compared with a group of controls (n = 23; age: M = 131 months SD = 13; IQ: M = 108 SD = 7). Reexamination has been performed after about one year. In ADHD EF deficits found at the beginning were decreased at the second testing after about one year when symptomatology had ameliorated. The boys with tic disorders revealed no differences compared to controls and no changes over time. The reduction of EF deficits with time in boys with ADHD is best explained by neuronal maturational processes, which are very intensive in the children with ADHD of this age.

Attention↗

[A treated case of facial tic caused by a psychological factor. Successful case approached by psychosomatic dentistry].

As the oral psychosomatic diseases of infants appear connected with a predisposition of their environment, environmental adjustment and psychotherapy on the basis of psychosomatic dentistry are necessary in the treatment. We encountered a 9-year-old boy who seemed to be suffering from a facial tic caused by psychological factors. An attempt of therapy on the basis of psychosomatic dentistry was made in this case. The results as follows were obtained: 1. Intake interview to a mother was carried out regarding a cause of the tic. It was shown that the attitude of the mother will bring up the child and her meddling as well as the stress of school life caused his facial tic. 2. With our guidance, the mother reconsidered her meddling attitude toward the patient. And she was able to handle the patient with a kind attitude. 3. After play therapy as preparation, catharsis was carried out on the patient, the tic decreased rapidly. 4. Finally a token economy method was used. 5. It was thought that sufficient correspondence on the basis of psychosomatic dentistry was beneficial in the therapy of patient with psychosomatic disease in pedodontics.

Child↗