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PubMed1996

Severe motor tics causing cervical myelopathy in Tourette's syndrome.

We describe two patients with Tourette's syndrome in whom severe motor tics involving the neck were complicated by cervical myelopathy. The first patient, a 21-year-old man, had complex tics consisting of violent twisting and extending movements of the neck preceded by an irresistible urge to produce the abnormal postures. Two years after onset of these tics, paraesthesias, sensory deficits up to the level of C4, and a gait disturbance developed. Neuroimaging studies confirmed compressive myelopathy, and the symptoms gradually improved with botulinum toxin injections in the posterior cervical muscles. The second patient, a 42-year-old man, had violent clonic tics consisting of thrusting head jerks. The repetitive flexion-extension neck movements had been present since the age 10 years. At age 23, he developed progressive weakness of all four extremities and bladder and sexual dysfunction. Myelography demonstrated cervical spinal canal stenosis with retrospondylosis from C3 through C5 and a Klippel-Feil malformation. After cervical decompression by C3-C5 laminectomies, his spinal cord symptoms improved temporarily. The tics, however, continued, and the neurologic deficits of cervical myelopathy progressed again after age 34. He did not benefit from a second operation. This report draws attention to the possibility that some tics can produce disabling compressive myelopathy. Early diagnosis and treatment of the movement disorder is essential in such cases.

Adult↗
PubMed2001

Differences in 99mTc-HMPAO brain SPET perfusion imaging between Tourette's syndrome and chronic tic disorder in children.

Early differential diagnosis between Tourette's syndrome and chronic tic disorder is difficult but important because both the outcome and the treatment of these two childhood-onset diseases are distinct. We assessed the sensitivity and specificity of brain single-photon emission tomography (SPET) perfusion imaging in distinguishing the two diseases, and characterized their different cerebral perfusion patterns. Twenty-seven children with Tourette's syndrome and 11 with chronic tic disorder (mean age 9.5 and 8.6 years, respectively) underwent brain SPET with technetium-99m hexamethylpropylene amine oxime (HMPAO). Visual interpretation and semiquantitative analysis of SPET images were performed. On visual interpretation, 22 of 27 (82%) of the Tourette's syndrome group had lesions characterized by decreased perfusion. The left hemisphere was more frequently involved. None of the children with chronic tic disorder had a visible abnormality. Semi-quantitative analysis showed that, compared with children with chronic tic disorder, children with Tourette's syndrome had significantly lower perfusion in the left lateral temporal area and asymmetric perfusion in the dorsolateral frontal, lateral and medial temporal areas. In conclusion, using the visual approach, brain SPET perfusion imaging is sensitive and specific in differentiating Tourette's syndrome and chronic tic disorder. The perfusion difference between the two groups, demonstrated by semi-quantitative analysis, may be related more to the co-morbidity in Tourette's syndrome than to tics per se.

Adolescent↗
PubMed2004

Treatment of phonic tics in patients with Tourette's syndrome using botulinum toxin type A.

We assessed the effect of botulinum toxin type A (BTX-A) on phonic tics in patients with Tourette's syndrome. A total of 30 patients received 2.5 IU BTX-A (BOTOX; Allergan) in both vocal cords. All patients were assessed after 15 days and then 4 times over a 12-month period. At each visit the following data were collected: phenomenology of tics, global impression of changes by physician and patient, number of BTX-A injections given, interval between injections, time to response, duration of response, presence of post-injection hypophonia and side effects, presence of premonitory sensory tic component, and interference with social life and work or school activities. Vocal tics improved after treatment in 93% patients, with 50% being tic-free. Mean response time was 5.8 days, and mean duration of response was 102 days. Quality of life improved, and premonitory experiences dropped from 53% to 20%. Hypophonia was the only side effect of note (80% of patients). BTX-A is an effective and safe treatment for phonic tics associated with Tourette's syndrome.

Activities of Daily Living↗
PubMed1986

A simple and a complex tic (Gilles de la Tourette's syndrome): their response to EEG sensorimotor rhythm biofeedback training.

This study presents a clinical treatment regime for the treatment of tic manifestation, both simple and complex. The response of a case of simple tic and a case of complex tic (Gilles de la Tourette's syndrome) to EEG sensorimotor rhythm biofeedback training are presented. Specifically, the simple and the complex tic, both of long duration, were eliminated via this EEG biofeedback training procedure. It is hypothesized that this exercising of the sensorimotor cortex resulted in increased activation of this cerebrocortical subsystem and was reflected in increased voluntary muscle control and a heightened threshold for random motor discharge, resulting in the elimination of both tics as in the response of cases of epilepsy with motor involvement to EEG sensorimotor rhythm biofeedback training. The additional psychophysiologic sequelae of the complex tic--attention deficit disorder--remediated in the manner of the response of learning-disabled to EEG sensorimotor rhythm biofeedback training.

Adolescent↗
PubMed2006

Tic disorders in childhood.

Despite our familiarity with tic disorders, their management remains challenging. The difficulty seldom relates to the diagnosis, which is usually based on the history, but rather in dealing with the impact of the tics and multiple comorbidities. The child and family must be educated and must be taught how to facilitate a positive attitude among others who come in contact with the patient. Knowledge and acceptance of the tics by other family members, peers, teachers, and coaches often become central to the child's well-being. Although many children can be managed without pharmacotherapy, medications are often central to achieving success. The goals of therapy and the appropriate choice of medication must be individualized. We are frequently challenged not only by the wide array of medication choices available but also by the decision of which symptom to treat first. The tics, despite being flamboyant, are often viewed by the child and family as less disabling than co-occurring conditions, such as attention-deficit hyperactivity disorder, obsessive-compulsiveness, or rage. Even when tics are the major issue, patients must define their goals for therapy. It is unreasonable to expect a complete resolution of tics and safety usually rules in favor of better tolerated but potentially less efficacious therapies. Developing strategies to minimize the lifelong impact of the multiple components of Tourette syndrome is an essential aspect of the care of these patients.

Adolescent↗
PubMed2003

Behavioral activity associated with onset in chronic tic and habit disorder.

Seventy-six people (aged 18-62 years) diagnosed with either a chronic tic disorder or a habit disorder, entering a treatment study, kept a baseline daily diary for at least ten days, noting tic frequency and activity at time of onset. Together with an evaluator, participants completed a form ranking three high-risk activities where the tic or habit was likely to appear, and three low-risk activities where the tic was absent or barely present. Subjective appraisals distinguishing the two types of activities were also elicited and their relevance to the tic was measured on a seven-point scale using an adaptation of Kelly's repertory grid technique. Overall, the most frequent high-risk and low-risk activities were, respectively, passive attendance and physical activity. There were, however, significant differences in types of high-risk activities amongst the tic and habit disorders. Conversely, appraisals of the high-risk activities seemed to center on negative evaluations of tenseness, boredom, dissatisfaction, and disinterest.

Adolescent↗
PubMed1999

Therapeutic plasma exchange and intravenous immunoglobulin for obsessive-compulsive disorder and tic disorders in childhood.

BACKGROUND: In children, exacerbations of tics and obsessive symptoms may occur after infection with group A beta-haemolytic streptococci. If post-streptococcal autoimmunity is the cause of the exacerbations, then children might respond to immunomodulatory treatments such as plasma exchange or intravenous immunoglobulin (IVIG). We studied whether plasma exchange or IVIG would be better than placebo (sham IVIG) in reducing severity of neuropsychiatric symptoms. METHODS: Children with severe, infection-triggered exacerbations of obsessive-compulsive disorder (OCD) or tic disorders, including Tourette syndrome, were randomly assigned treatment with plasma exchange (five single-volume exchanges over 2 weeks), IVIG (1 g/kg daily on 2 consecutive days), or placebo (saline solution given in the same manner as IVIG). Symptom severity was rated at baseline, and at 1 month and 12 months after treatment by use of standard assessment scales for OCD, tics, anxiety, depression, and global function. FINDINGS: 30 children entered the study and 29 completed the trial. Ten received plasma exchange, nine IVIG, and ten placebo. At 1 month, the IVIG and plasma exchange groups showed striking improvements in obsessive-compulsive symptoms (mean improvement on children's Yale-Brown obsessive compulsive scale score of 12 [45%] and 13 [58%], respectively), anxiety (2.1 [31%] and 3.0 [47%] improvement on National Institute of Mental Health anxiety scale), and overall functioning (2.9 [33%] and 2.8 [35%] improvement on National Institute of Mental Health global scale). Tic symptoms were also significantly improved by plasma exchange (mean change on Tourette syndrome unified rating scale of 49%). Treatment gains were maintained at 1 year, with 14 (82%) of 17 children "much" or "very much" improved over baseline (seven of eight for plasma exchange, seven of nine for IVIG). INTERPRETATION: Plasma exchange and IVIG were both effective in lessening of symptom severity for children with infection-triggered OCD and tic disorders. Further studies are needed to determine the active mechanism of these interventions, and to determine which children with OCD and tic disorders will benefit from immunomodulatory therapies.

Adolescent↗
PubMed1997

Increased prevalence of the seven-repeat variant of the dopamine D4 receptor gene in patients with obsessive-compulsive disorder with tics.

The polymorphism characterized by a varying number of 48 bp repeats (VNTR) in the dopamine D4 receptor (DRD4) gene was examined in 61 obsessive-compulsive disorder (OCD) probands with and without tics. Most of the OCD patients with tics showed at least one copy of the 7-fold variant compared to those affected subjects without tics (91 vs. 48%, respectively, Yates corrected chi2 = 5.54, P = 0.018). Similarly, a higher number of copies of this common variant were detected in the group of probands displaying tics compared to those OCD's without tics (Yates corrected chi2 = 4.66, P = 0.03). Our study suggests that the seven-repeat allele of the DRD4 gene could be a factor in the phenotypic variance of tics among OCD individuals.

Alleles↗
PubMed2003

Tics and developmental stuttering.

BACKGROUND: Developmental stuttering affects 1% of the population but its cause remains unclear. Recent PET studies of metabolism in the central nervous system suggest that it may be related to dysfunction in the basal ganglia or its connections with regions of the cortex associated with speech and motor control. OBJECTIVE: To determine the presence and characteristics of involuntary movements (IMs) in people who stutter and to investigate the hypothesis that these movements may be of a very similar nature to the IMs seen in patients with movement disorders due to basal ganglia dysfunction. METHODS: Sixteen adults with developmental stuttering and 16 controls matched for sex and age were audio-videotaped while freely speaking 300 words in conversation and reading aloud 300 words. The audio data was inspected for dysfluencies and the video data was scrutinised for the presence and characteristics of IMs. RESULTS: Subjects who stuttered produced more IMs than controls during free speech (354 vs 187, p<0.05) and reading (297 vs 47, p<0.001). Most of the IMs in both groups were tics, with a greater number of both simple and complex motor tics (CMTs) in subjects who stuttered. CMTs were more frequent than simple motor tics in those who stuttered, but not in controls. The combination of repetitive eye blink followed by prolonged eye closure was found exclusively in the stuttering group, as were simple tics consisting of eyebrow raise or jaw movement. Dystonia in the form of blepharospasm was identified in a small number of subjects who stuttered. Choreic movements were not associated with stuttering. CONCLUSIONS: Developmental stuttering is associated with the presence of IMs that are predominantly simple and CMTs. This association suggests that tics and stuttering may share a common pathophysiology and supports the view that, in common with tics, stuttering may reflect dysfunction in the basal ganglia or its immediate connections.

Adolescent↗
PubMed1989

Pain in Gilles de la Tourette syndrome and related tic disorders.

Although pain is not generally recognized as a symptom of tic disorders, we have seen a number of patients in whom this was a prominent feature, at times even the symptom of greatest concern. The commonest pain complaints are those arising from the actual performance of a tic. Most often this follows directly from the discomfort produced by sudden or repeated extreme exertion. Here the origin of the pain is usually musculoskeletal, though rare examples of neuropathic pain may occur. Pain also may arise from striking or being struck by a moving body part involved in large amplitude tics. Other related painful acts include deliberate self-injury and pain inflicted upon others. A second major category is represented by a smaller number of patients who complain of pain during voluntary efforts to suppress their tics. Finally, there are patients who obtain relief from tics while experiencing pain, to such an extent that they will deliberately provoke pain to obtain its benefit. We feel that pain should be recognized as a common complaint, and occasionally a source of significant disability, in patients with tics.

Adolescent↗
PubMed2006

Comorbid behavioural problems in Tourette's syndrome are positively correlated with the severity of tic symptoms.

OBJECTIVE: We studied the comorbid behavioural and mood problems in children with non-psychiatric Tourette's syndrome (TS) and their relationship with severity of tic disorder. METHOD: Sixty-nine TS children and 69 healthy controls were assessed by Child Behavior Checklist (CBCL) and Yale Global Tic Severity Scale (YGTSS). The relationships between behavioural problems and severity of tic symptoms were analysed statistically by comparison, correlation and multiple linear regression. RESULTS: Tourette's syndrome patients scored significantly lower (p<0.01) on the CBCL competency subscales and total score, and higher on all behavioural problem subscales and total score (p<0.01). Expectedly, the TS children had lower social competence than normal children. Among the TS children, the severity of tic symptoms is positively correlated with the severity of overall impairment in school and social competence. When the behavioural and mood problems commonly associated with TS were studied in detail, we found that delinquent behaviour, thought problems, attention problems, aggressive behaviour and externalizing are positively correlated with severity of tic symptoms. CONCLUSION: The findings indicated that children with TS-only also had a broad range of behavioural problems, and some of these were related to the severity of tic symptoms.

Adolescent↗
PubMed2005

Association of common cold with exacerbations in pediatric but not adult patients with tic disorder: a prospective longitudinal study.

Cross-sectional data and case studies suggest a temporal relationship between fluctuations in tic severity and preceding infections. In this study, we aimed to examine this possible relationship in a prospective longitudinal design. Two groups of tic disorder patients were included, a pediatric group between 7 and 15 years of age (n = 20), and an adult group over 15 years of age (n = 41). During a 24-week period, participants were asked to fill out weekly self questionnaires regarding the presence of tic exacerbations and the experience of the common cold. In addition, 6 throat swabs were taken at monthly intervals and cultured for streptococci; also, 3 serial serum assessments of streptococcal antibodies were performed at 8-week intervals. In the pediatric group, our results indicated a strong association between the self report of a common cold and a symptom exacerbation 4 weeks later (Odds ratio = 4.685; p = 0.001). In the adult group, we found no association between reports of common cold and tic exacerbations. Association with streptococcal infections could not be determined owing to the limited number of observed streptococcal infections. Thus, this study points to a hitherto unknown association of common viral infections with tic exacerbations in children, which may support the involvement of immune dysregulation in tic disorders.

Adolescent↗
PubMed1995

Risperidone treatment of children and adolescents with chronic tic disorders: a preliminary report.

OBJECTIVE: The purpose of this trial was to investigate the short-term safety and efficacy of risperidone in the treatment of chronic tic disorders in children and adolescents. METHOD: This was an 11-week open-label trial and included seven subjects (five boys and two girls) with a mean age of 12.9 +/- 1.9 years. The sample included five patients with Tourette's syndrome and two with chronic motor tic disorder. The children were seen at baseline and for two follow-up visits. Three children had a comorbid diagnosis of obsessive-compulsive disorder (OCD). RESULTS: Clinical response, as measured by the Yale Global Tic Severity Scale and the Children's version of the Yale-Brown Obsessive Compulsive Scale, revealed a statistically significant reduction in tic scores ranging from 26% [corrected] to 66%. One of three children with comorbid OCD showed substantial improvement; the other two subjects showed no change. The most frequent side effect was weight gain, which ranged from 8 to 14 lb. CONCLUSIONS: Risperidone, a neuroleptic with both serotonin- and dopamine-blocking properties, appears to be effective in reducing tic frequency and intensity in children and adolescents with chronic tic disorders.

Adolescent↗
PubMed2001

Gilles de la Tourette's syndrome with and without obsessive-compulsive disorder compared with obsessive-compulsive disorder without tics: which symptoms discriminate?

Stereotyped repetitive behaviors occur in Gilles de la Tourette's Syndrome (GTS) and obsessive-compulsive disorder (OCD). The present study was undertaken to compare the distribution of obsessive-compulsive and Tourette-related impulsive behaviors in GTS with (+) OCD, GTS without (-) OCD, tic-free OCD, and control subjects. Fourteen GTS + OCD, 18 GTS-OCD, 21 OCD-tic, and 29 control subjects were evaluated using a semistructured interview designed to assess GTS and OCD-related repetitive behaviors. Each reported item was evaluated on the presence of anxiety and goal-directedness. This information was subsequently used to define whether the repetitive behavior was an (anxiety-related) obsession or compulsion, or a (non-anxiety-related) OC-like behavior, impulsion. GTS + OCD subjects reported more overall Tourette-related impulsions than OCD-tic subjects, i.e., more mental play, echophenomena, and touching behaviors but similar frequencies of typical obsessive-compulsive behaviors. Further, GTS + OCD subjects exhibited more overall repetitive behaviors than GTS-OCD subjects, i.e., more Tourette-related impulsions as well as more obsessive-compulsive behaviors. The distribution of symptoms is similar in GTS with and without OCD, and differs from tic-free OCD. These differences suggest that GTS with OCD constitutes a form of GTS, not of OCD, although the possibility that GTS + OCD patients constitute a subgroup distinct from GTS and from OCD can not be excluded by this phenomenological study. Specific non-anxiety-related impulsions seem to discriminate between GTS and OCD-tic individuals. These impulsions possibly reflect differences in underlying mechanisms between GTS and OCD-tics.

Adult↗
PubMed1997

Differences in clinical characteristics between Tourette syndrome patients with and without 'generalized tics' or coprolalia.

The purpose of this study is to examine whether there are differences in clinical characteristics between Tourette syndrome (TS) patients with and without 'generalized tics' (GT) which involve the entire body, and/or coprolalia. Subjects were 64 patients (55 males and 9 females, mean age, 17.4 +/- 7.2 years) who visited Tokyo University's outpatient clinic of neuropsychiatry from 1974 to 1993 and who met criteria for Tourette's disorder of DSM-III-R. Data on clinical characteristics, including tic symptoms and courses of their development, complications and developmental histories, treatment and severity, were collected by systematic chart review of all subjects. Tourette syndrome patients with 'generalized tics' tended to show multiple complex vocal tics more frequently than TS patients without GT. Tourette syndrome patients with coprolalia tended to show significantly higher rates of copropraxia, echolalia, and 'cleaning/washing' compulsion than did the TS patients without coprolalia. Tourette syndrome patients with both GT and coprolalia were classified as the severest group in terms of tic symptoms and social impairment. Tourette syndrome patients who had neither of these morbidities were classified into the mildest group in all aspects. Generalized tics and coprolalia seemed to indicate the severest end of the TS spectrum and seemed to be related with a need of intensive treatment.

Adolescent↗
PubMedDate not supplied

Carbamazepine-induced tics.

A variety of movement disorders are known to occur in association with carbamazepine (CBZ) therapy in adults and children, but development of tics has been described infrequently and only in patients with underlying Tourette's syndrome or other movement disorders. We report 3 children with epilepsy who developed facial motor tics after initiation of CBZ for complex partial seizures. All 3 had documented CBZ blood levels in the therapeutic range at the time, and none had other symptoms or signs of clinical intoxication. Neurologic examinations were normal in 2 and showed developmental delay of expressive language in the third. Brain imaging was normal in all. After development of the tics in 2, CBZ was continued at the same or higher dose, and the tics abated and then ceased spontaneously < or = 6 months. In the third child, the tics ceased after CBZ discontinuation. These cases demonstrate that CBZ can induce simple motor tics in children. These idiosyncratic reactions may be transient and do not always necessitate drug discontinuation.

Adolescent↗
PubMed2001

Elevated D8/17 expression on B lymphocytes, a marker of rheumatic fever, measured with flow cytometry in tic disorder patients.

OBJECTIVE: Elevated D8/17 expression on B lymphocytes is a known susceptibility marker of rheumatic fever. Previous studies have reported higher than usual D8/17 expression on B lymphocytes of patients with tic disorders. The purpose of this study was to assess D8/17 expression on B lymphocytes of tic disorder patients by using an objective method in which no operator variability was involved. METHOD: D8/17 expression on B lymphocytes was assessed with flow cytometry by using an immunoglobulin M (IgM) monoclonal D8/17-specific antibody in an unselected group of Dutch patients with tic disorders (N=33) and healthy volunteers (N=20). Binding of this monoclonal antibody was compared with binding of an irrelevant IgM monoclonal antibody, and the shift in mean fluorescence intensity of the D8/17-specific antibody compared to that of the irrelevant IgM monoclonal antibody was used as a measure of D8/17 overexpression. For the patients, Yale Global Tic Severity Scale scores were used to assess disease severity. RESULTS: D8/17 overexpression in the patient group (mean=16.8 arbitrary units, SD=30.5) was significantly higher than in the comparison group (mean=3.2, SD=3.0). A significant minority of the patients (N=13, 39.4%), however, had levels of D8/17 overexpression within the range of that of the healthy comparison subjects. Flow cytometric analysis did not indicate a separate subpopulation of D8/17-positive B cells. CONCLUSIONS: These data confirm the utility of D8/17 B cell overexpression as a peripheral blood marker in patients with tic disorders and are compatible with a streptococcus-related pathogenesis for at least a subgroup of patients with tic disorders.

Adolescent↗
PubMed2002

Assessment and treatment of vocal tics associated with Tourette's syndrome.

A functional analysis suggested that a young man's vocal tics were maintained by automatic reinforcement. A preference assessment was conducted to identify stimuli that effectively competed with the occurrence of vocal tics. When used as components of a reinforcement-based intervention, however, these stimuli were ineffective at reducing the occurrence of vocal tics. Observations conducted in a naturalistic context led to the hypothesis that variations in tics were associated with body positioning. Thus, an additional analysis was conducted to determine if vocal tics occurred less when the participant was lying down versus when he was seated upright. Results suggested that a combination of procedures might be useful in developing idiosyncratic interventions for automatically reinforced problem behavior, such as vocal tics.

Adult↗