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Showing papers on "Tourette syndrome published in 1991"


Journal ArticleDOI
02 Oct 1991-JAMA
TL;DR: The results suggest the A1 allele of the DRD2 gene is associated with a number of behavior disorders in which it may act as a modifying gene rather than as the primary etiological agent.
Abstract: Objective. —The A1 allele of the Taq I polymorphism of the dopamine D 2 receptor ( DRD2 ) gene has been earlier reported to occur in 69% of alcoholics, compared with 20% of controls. Other research has reported no significant difference in the prevalence of the A1 allele in alcoholics vs controls and no evidence that the DRD2 gene was linked to alcoholism. We hypothesized that these seemingly conflicting results might be because increases in the prevalence of the A1 allele may not be specific to alcoholism. Thus, we examined other disorders frequently associated with alcoholism or those believed to involve defects in dopaminergic neurotransmission. Design. —Case comparison study. To minimize the effect of racial differences in gene frequencies, the study was restricted to non-Hispanic whites. Setting. —Ambulatory and hospitalized patients. Results. —Among all known controls (n = 314), 77 (24.5%) carried the A1 allele. Of the 69 controls known not to be alcoholics, 10 (14.5%) carried the A1 allele. The prevalence of the A1 allele was significantly increased in patients with Tourette's syndrome (44.9%, n = 147), attention deficit hyperactivity disorder (46.2%, n = 104), autism (54.5%, n = 33), alcoholism (42.3%, n = 104), and posttraumatic stress disorder (45.7%, n = 35). After correction for multiple comparisons (requiring P Conclusion. —These results suggest the A1 allele of the DRD2 gene is associated with a number of behavior disorders in which it may act as a modifying gene rather than as the primary etiological agent. ( JAMA . 1991;266:1793-1800)

593 citations


Journal ArticleDOI
TL;DR: Results indicate that clonidine is more effective than placebo in reducing some of the tic and other behavioral symptoms associated with Gilles de la Tourette's syndrome.
Abstract: • The safety and effectiveness of clonidine hydrochloride (3 to 5 μg/kg per day) were evaluated in 47 subjects with Gilles de la Tourette's syndrome, aged 7 to 48 years. Twenty-four subjects were randomly assigned to clonidine treatment and 23 to placebo. Forty subjects (21 given clonidine and 19 placebo) successfully completed the 12-week, double-blind clinical trial. Clinical ratings of tic severity improved for both groups. The magnitude of response was greater in the group receiving clonidine. Clinician-rated measures of motor tic severity, the degree to which the tics are "noticeable to others," motor tic counts from videotaped interviews, and parent-rated measures of impulsivity and hyperactivity were the most responsive to clonidine treatment. These results indicate that clonidine is more effective than placebo in reducing some of the tic and other behavioral symptoms associated with Gilles de la Tourette's syndrome.

317 citations


Journal Article
TL;DR: The rates of TS, CT, and OCD in the total sample of biological relatives of TS probands were significantly greater than in the relatives of controls, providing further evidence that OCD is etiologically related to TS.
Abstract: Previous studies have demonstrated that Gilles de la Tourette syndrome (TS) is a familial disorder and that chronic tics (CT) and obsessive compulsive disorder (OCD) appear to be etiologically related to the syndrome. In the present study we report the results from a study of 338 biological relatives of 86 TS probands, 21 biologically unrelated relatives of adopted TS probands, and 22 relatives of normal subjects. The 43 first-degree relatives of the adopted TS and normal probands constituted a control sample. The rates of TS, CT, and OCD in the total sample of biological relatives of TS probands were significantly greater than in the relatives of controls. In addition, the morbid risks of TS, OCD, and CT were not significantly different in families of probands with OCD when compared to relatives of probands without OCD. These findings provide further evidence that OCD is etiologically related to TS.

288 citations


Journal ArticleDOI
01 Jan 1991-Medicine
TL;DR: The definition, characteristics, pathophysiology, and treatment of tic disorders with a major emphasis on Tourette syndrome are discussed, with current evidence supporting a pathophysiologic mechanism involving synaptic neurotransmission, with the dopaminergic system as a primary candidate.

181 citations


Journal ArticleDOI
TL;DR: The rare appearance of the different dyskinesias and normal movements under similar circumstances during sleep could be a result of common effects on the generator systems or changes in the excitability of the final common motor pathway.
Abstract: • The effect of sleep on the involuntary movements or dyskinesias in Parkinson's disease, Huntington's disease, primary and secondary torsion dystonia, and Gilles de la Tourette syndrome was studied in a total of 52 patients and 10 normal subjects using video electroencephalographic telemetry. Movements typical of the wake pattern were seen occasionally during unequivocal sleep in all but two completed studies, and in each condition reappeared under similar circumstances. The movements were most likely to occur after awakenings or lightenings of sleep, or in stage one sleep. The movements were very rare during the deeper phases of sleep. Those movements that occurred during sleep without awakenings were usually preceded by arousal phenomena and, rarely, by sleep spindles or slow waves. The control group showed normal "semipurposeful" movements under the same conditions during sleep. The rare appearance of the different dyskinesias and normal movements under similar circumstances during sleep could be a result of common effects on the generator systems or changes in the excitability of the final common motor pathway.

129 citations


Journal ArticleDOI
TL;DR: It is proposed that some types of PDD are inherited in the same fashion and by the same gene, which would explain the similarity of symptoms, frequent evolution ofPDD into TS, the apparent recessive inheritance of PD D despite no increase in consanguinity, the high frequency of behavior problems in the relatives of PDd----TS patients and the serotonin abnormalities.
Abstract: Children with autism or pervasive developmental disorder (PDD) and Tourette syndrome (TS) share a number of symptoms. Forty-one cases have been reported in which PDD patients subsequently developed TS. We term this PDD----TS. We describe an additional 16 such patients plus 3 families where a close relative of a TS proband had autism. There was a high frequency of alcoholism, drug abuse, obsessive-compulsive, and other behavior disorders in the relatives of these patients. This frequency was virtually identical to that observed in relatives of individuals with TS only. We suggest there is an intimate genetic, neuropathologic relatedness between some cases of PDD and TS. Many observations have led us to suggest that the genetic defect in TS may be a mutation of tryptophan oxygenase and that TS is inherited as a semidominant semirecessive trait, i.e., homozygosity for a common gene which shows some expression in the heterozygous state. We propose that some types of PDD are inherited in the same fashion and by the same gene. This would explain the similarity of symptoms, frequent evolution of PDD into TS, the apparent recessive inheritance of PDD despite no increase in consanguinity, the high frequency of behavior problems in the relatives of PDD----TS patients and the serotonin abnormalities.

97 citations


Journal Article
TL;DR: An overview of the methods and progress of the groups centered at Yale University and Erasmus University in excluding linkage from a large portion of the genome is presented.
Abstract: Gilles de la Tourette syndrome is a neuropsychiatric disorder with an autosomal dominant mode of inheritance and reduced penetrance at a single genetic locus. Several research groups have genetic linkage studies underway to detect the chromosomal location of the gene that predisposes for this disorder. Strong and clear evidence of linkage has not yet been produced for Tourette syndrome. This paper presents an overview of the methods and progress of the groups centered at Yale University and Erasmus University in excluding linkage from a large portion of the genome. Our labs have screened 228 genetic marker loci for linkage with a gene for this disorder in a series of affected families in the United States, Canada, The Netherlands, and Norway. More than 50% (and perhaps as much as 66%) of the autosomal genome has now been excluded on the assumption that genetic heterogeneity is not an important factor in the Tourette syndrome pedigrees pooled for this summary.

81 citations


Journal ArticleDOI
TL;DR: It is shown that pharmacological manipulation of the endogenous opioid system does influence symptoms of Tourette's syndrome and the Trail Making B test best discriminated the treatments.
Abstract: To investigate the effect of drugs acting on the endogenous opioid system, we studied 10 adults with Tourette's syndrome who received propoxyphene hydrochloride (260 mg/day), naltrexone hydrochloride (50 mg/day), and placebo in a double-blinded, randomized clinical trial. Using a self-report scale (Tourette's Syndrome Symptom List), subjects noted a significant (p less than 0.04) lessening of tics after treatment with naltrexone when compared with placebo. An improvement in performance on the Trail Making B test, a measure of attention and visuomotor sequencing and planning, occurred after receiving naltrexone when compared with placebo (p less than 0.08) or propoxyphene (p less than 0.02). The Trail Making B test best discriminated the treatments (p less than 0.02, analysis of variance). No other treatment effects were observed for several other measures of tic severity, attentional ability, or obsessive-compulsive symptoms. Our findings indicate that pharmacological manipulation of the endogenous opioid system does influence symptoms of Tourette's syndrome.

70 citations


Book
01 Aug 1991
TL;DR: This book describes the application of single photon emission tomography (SPET) to neuroactivation imaging in particular and neuroimaging in general and results are given and discussed on the basis of clinical material and case histories.
Abstract: This book describes the application of single photon emission tomography (SPET) to neuroactivation imaging in particular and neuroimaging in general. Protocols for SPET, neuroactivation and neuroimaging are described in detail and results are given and discussed on the basis of clinical material and case histories. Normal functional anatomy is correlated with data from MRI. High resolution cerebral blood flow (CBF) imaging is described with the use of SPET technology. Split dose CBF imaging is also mentioned, for activation studies in a variety of normal and diseased states which include the dementias (AIDS, Alzheimer's disease and multi-infarction dementia), cerebrovascular disease, depression, schizophrenia, obsessive compulsive disorders, movement disorders (Parkinson's disease, Gilles de la Tourette syndrome, Huntingdon's chorea, Sydenham's chorea), epilepsy and tumours. Examples of motor, frontal and visual activation studies are also given.

64 citations


Journal ArticleDOI
TL;DR: Evidence from the histories of the patients and their relatives combined with other reports of case of cooccurrence of TS and autism provides support for the hypothesis that TS may be responsible for cases of coocurrence of the disorders, contributes significantly to the etiological heterogeneity of autistic disorder.
Abstract: The histories of 10 children with autistic disorder or pervasive developmental disorder (PDD) cooccurring with familial Tourette syndrome (TS) are presented. Evidence from the histories of the patients and their relatives combined with other reports of cases of cooccurrence of TS and autism provides support for the hypothesis that TS may be responsible for cases of coocurrence of the disorders, contributes significantly to the etiological heterogeneity of autistic disorder and that a portion of cases of autism may actually be a result of homozygosity for the TS gene. In addition, the presence of affective disorders and autistic-like syndromes or mild disturbances of social relatedness in some of the pedigrees suggests the hypothesis that TS may be responsible for a subgroup of families with cooccurring affective and autistic disorders and for some cases of familial aggregation of autism-PDD.

60 citations


Journal ArticleDOI
TL;DR: Performance on the Wisconsin Card Sorting Test was correlated with ratings of OC characteristics, but not with other TS symptoms, and it was speculated that different symptoms associated with TS may have different neuroanatomic substrates.
Abstract: The relationship between obsessive-compulsive (OC) characteristics and performance on a test sensitive to frontal lobe function (Wisconsin Card Sorting Test) was examined in a sample of 100 patients between the ages of 6 and 18 years. All patients met DSM-III-R criteria for Tourette syndrome (TS), confirmed by a neurologist or psychiatrist. Performance on the Wisconsin Card Sorting Test was correlated with ratings of OC characteristics, but not with other TS symptoms. This relationship was maintained even when Full-Scale IQ and the total number of Tourette symptoms were controlled. The effect could not be attributed to medication. These findings were interpreted in the context of models of basal ganglia-cortical associations. It was speculated that different symptoms associated with TS may have different neuroanatomic substrates.

Journal ArticleDOI
TL;DR: This study examined performance on a battery of neuropsychological tests in a sample of 28 patients with Tourette's syndrome and found neuroPsychological performance was mildly below average.
Abstract: This study examined performance on a battery of neuropsychological tests in a sample of 28 patients with Tourette's syndrome (TS). Test scores were converted to age-corrected T-scores to control for the effect of age on test performance. The frequency of abnormal test performances was variable, but more frequent on motor and sensory tasks. Symptom severity as measured by the Tourette Syndrome Global Scale was inversely related to neuropsychological performance. In general, neuropsychological performance was mildly below average. The pattern of performance was similar to previous studies of TS patients.


Journal ArticleDOI
TL;DR: A brief overview of the safety and efficacy of psychotropic drugs and the issues associated with their use in clinical settings is presented.
Abstract: During the past two decades psychopharmacologists have made considerable strides in establishing the safety and efficacy of psychotropic drug therapy for childhood behavior disorders. Most of the research has focused on children with disruptive behavior disorders, autism, or mental retardation, but more recently other disorders such as depression, obsessive compulsive disorder, separation anxiety (school refusal), and Tourette syndrome are also receiving attention. Psychopharmacotherapy has often been a matter of controversy, with most issues pertaining to either the appropriateness of medication (e.g., rationales for treatment, alternative interventions, toxicity, iatrogenic effects) or inadequacies of clinical management (e.g., availability of services, drug assessment procedures, limitations of research). This article presents a brief overview of the safety and efficacy of psychotropic drugs and the issues associated with their use in clinical settings.

Journal ArticleDOI
TL;DR: In a cohort of five patients from the Middle East with the Gilles de la Tourette syndrome, family history of a tic disorder or the Gille de la tourette syndrome was positive in three cases.
Abstract: In a cohort of five patients from the Middle East with the Gilles de la Tourette syndrome, family history of a tic disorder or the Gilles de la Tourette syndrome was positive in three cases. In one of these there was a multiply affected pedigree spanning six generations. The phenomenology of the syndrome is the same as that described in Western reports. The familial pattern of inheritance and cross-cultural similarity emphasise the biological factors in the aetiology of the syndrome.


Journal ArticleDOI
TL;DR: This is the first reported case of childhood-onset Tourette syndrome with adult onset Huntington disease, and discovery of other cases with both disorders may provide clues to the pathophysiology of both conditions.
Abstract: A 40-year-old man with childhood-onset Tourette syndrome (TS) developed Huntington disease (HD). We believe this to be the first reported case of childhood-onset TS with adult onset HD. Discovery of other cases with both disorders may provide clues to the pathophysiology of both conditions.

Journal ArticleDOI
TL;DR: The author theorizes that abnormal discharges in the frontal lobes comprise the "final common dysfunction" that results in numerous phenomena labelled Tourette syndrome, and can account more readily for heterogeneity of clinical presentations, the numerous "dual diagnosis" cases, and differential response to medication.
Abstract: The etiology of Tourette Syndrome (TS) according to prevailing views is unknown; there is evidence for both familial and sporadic cases. The author theorizes that abnormal discharges in the frontal lobes comprise the "final common dysfunction" that results in numerous phenomena labelled Tourette syndrome. Facial, vocal, and other motor symptoms of TS are catalogued in parallel with facial, vocal, and body movements that occur during frontal lobe seizures. The variety of etiologies that cause frontal lobe seizures--when applied to TS--can account more readily for heterogeneity of clinical presentations, the numerous "dual diagnosis" cases, and differential response to medication than can a single-gene theory.

Journal ArticleDOI
TL;DR: Two patients with Gilles de la Tourette syndrome showed tics triggered by external stimuli, of significance to the aetiology of G TS and in particular the relationship between GTS and the startle response.
Abstract: Two patients with Gilles de la Tourette syndrome (GTS) showed tics triggered by external stimuli. This unusual feature is of significance to the aetiology of GTS and in particular the relationship between GTS and the startle response.

Book
01 Jan 1991
TL;DR: Innate specialization for emotion - temperament differences in children with early left versus right brain damage, R.Nass and D.Bregman attention-deficit hyperactivity disorder - family-genetic risk factors and comorbidity.
Abstract: Innate specialization for emotion - temperament differences in children with early left versus right brain damage, R.Nass and D.Koch overfocusing - an apparent subtype of attention deficit-hyperactivity disorder, M.Kinsbourne variability in the clinical presentation of autism - issues of diagnosis and treatment in the preschool years, D.A.Allen assessment of psychiatric status in the child with cerebral dysfunction - the issue of dual diagnosis, J.D.Bregman attention-deficit hyperactivity disorder - family-genetic risk factors and comorbidity, B.Biederman et al from tics to Tourette Syndrome, G.E.Solomon preschool children with inadequate language acquisition - implications for differential diagnosis and clinical management, I.Rapin and D.A.Allen the role of augmentative communication in impaired language acquisition, N.Amir et al neuropsychological assessment of children with developmental disabilities, L.Wainwright et al developmental dyslexia - an update, R.Nass developmental dyscalculia, R.S.Shaley and R.Wertman-Elad.


Journal ArticleDOI
TL;DR: Findings are in accord with recent evidence of little or no EEG abnormality in Tourette Syndrome patients as compared to normals.
Abstract: EEG abnormality has been reported in Gilles de la Tourette Syndrome but not confirmed in later studies. We carried out computerized EEG frequency analysis in 30 patients with the disorder, using Nicolet Pathfinder II frequency analysis software, versions 1.2 and 3.1 EEG was recorded from 01-A1+A2, 02-A1+A2, Fz-A1+A2, F7-C3, F8-C4, T5-01, and T6-02 in Tourette Syndrome patients and controls. Controls were taking no medications, and drug therapy for Tourette Syndrome had been stopped or not yet initiated in the patient group. Modal alpha frequency (MAF), maximal alpha frequency (MxAF), and spectral edge frequency (SEF) was measured in occipital and frontal derivations in 24 patients and controls. Left frontal (MOLF) and right frontal (MORF) mobility was calculated in F7-C3 and F8-C4 in 21 patients and controls. No significant differences were found between Tourette Syndrome patients and controls by two-tailed t-test. These findings are in accord with recent evidence of little or no EEG abnormality in Tourette Syndrome patients as compared to normals.

Journal ArticleDOI
TL;DR: 5. Jefferson JW, Greist JH, Baudhuin M, et al, Lithium and tryptophan augmentation in clomipramine-resistant obsessive-compulsive disorder.
Abstract: 5. Jefferson JW, Greist JH, Baudhuin M, et al. Lithium for obsessive-compulsive disorder? Psychosomatics 1984; 25: 493. 6. Rasmussen SA. Lithium and tryptophan augmentation in clomipramine-resistant obsessive-compulsive disorder. Am J Psychiatry 1984; 141: 1283-1285. 7. Feder R. Lithium augmentation of clomipramine. J Clin Psychiatry 1988; 49: 458. 8. Golden RN, Morris JE, Sack DA. Combined lithium-tricyclic treatment of obsessive-compulsive disorder. BioI Psychiatry 1988; 23: 181-185. 9. Eisenberg J, Asnis G. Lithium as an adjunct treatment in obsessive-compulsive disorder. Am J Psychiatry 1985; 142: 663. 10. Wernicke JF, Dunlop SR, Dornseif BE, et al. Low-dose fluoxetine therapy for depression. Psychopharmacol Bull 1988; 24: 183-188. II. Steiner W, Fontaine R. Toxic reaction following the combined administration of fluoxetine and L-tryptophan: five case reports. BioI Psychiatry 1986; 1067-1071.

Journal ArticleDOI
TL;DR: The phenomenology and aetiology of OCD will be reviewed, with particular emphasis placed on the proper pharmacological treatment of this sometimes crippling disorder.
Abstract: Within the past decade the field of psychiatry has rediscovered the neuropsychiatric syndrome of obsessive-compulsive disorder (OCD). Although excellently described over 150 years ago, for many years OCD was thought to be rare, untreatable, and to arise from hidden psychodynamic conflicts. All of these earlier ideas now appear to be wrong. Occurring in approximately 2% of adults, OCD consists of recurrent intrusive thoughts (obsessions) or senseless repetitive actions (compulsions). Although the aetiology of OCD remains unclear, recent neuro-imaging studies implicate the basal ganglia and frontal cortex as crucial structures in the pathogenesis of OCD. Genetic studies demonstrate a clear genetic component to OCD and an interesting link with chronic motor tics and the Gilles de la Tourette Syndrome. Although a true cure for the disorder remains elusive, most OCD symptoms respond well to treatment with 5HT reuptake inhibitors. The phenomenology and aetiology of OCD will be reviewed, with particular emphasis placed on the proper pharmacological treatment of this sometimes crippling disorder.

Journal ArticleDOI
TL;DR: There is a clear and strong association between obsessional thoughts and behaviours and GTS, and this is seen both in patients with GTS and in their family members.
Abstract: Georges Gilles de la Tourette first drew attention to the psychopathology of the Gilles de la Tourette Syndrome (GTS) in 1889 when he commented on the anxieties and phobias of his patients. In this paper he acknowledged the contribution of Guinon, who in 1886 had noted that "tiquers" nearly always had associated psychiatric disorders characterised by multiple phobias, arithmomania and agoraphobia. Since that time many types of psychopathology have been documented in association with GTS, including depression, anxiety, phobic disorder, hostility and aggression. However, the exact association between these disorders and GTS remains unclear. What is becoming increasingly evident is that there is a clear and strong association between obsessional thoughts and behaviours and GTS, and this is seen both in patients with GTS and in their family members. There have now been at least twenty investigations which have reported on this association, which is evident in clinic patients, epidemiological studies and family/pedigree populations. There have also been convincing arguments for the association being genetic.

Journal ArticleDOI
03 Apr 1991-JAMA

Journal ArticleDOI
TL;DR: Twenty-six children diagnosed with chronic tic disorders were studied for unexplained physical complaints and no significant correlation was found between the increased somatic complaints and the severity of anxiety, dysphoria, or movement disorder.


Journal ArticleDOI
TL;DR: The first English translations of their works referring to the psychopathology of Gilles de la Tourette syndrome are presented, comparing and contrasting their ideas to current concepts.
Abstract: Gilles de la Tourette gained eponymous fame when he described nine cases of multiple tics, coprolalia and echolalia, and later he, Guinon and Grasset were the first to document the psychopathology of the Gilles de la Tourette syndrome. In particular, they noted the association between obsessional thoughts and behaviours and the tic disorder. In this paper we present the first English translations of their works referring to the psychopathology, comparing and contrasting their ideas to current concepts.