Sociodemographic and clinical characteristics of children with tic disorders and behavioral problems: A real-world study and development of a prediction model.

BACKGROUND: Tic disorders (TD) are complex neuropsychiatric disorders frequently associated with a variety of comorbid problems, whose negative effects may exceed those of the tics themselves. In this study, we aimed to explore the sociodemographic and clinical characteristics of children with TD and behavioral problems, and develop a prediction model of behavioral problems based on the predictors under real-world conditions. METHODS: A hospital-based cross-sectional study was conducted on children with TD. Behavioral problems were surveyed using the Achenbach Child Behavior Checklist (CBCL). Sociodemographic information was collected from face-to-face interviews using an electronic questionnaire administered during the initial ambulatory visit. Clinical data were collected from medical records, and quality control was performed. The sociodemographic and clinical characteristics of patients with and without behavioral problems were statistically compared, and a nomogram prediction model was developed based on multivariate logistic regression analysis. The discriminatory ability and clinical utility of the nomogram were assessed by concordance index (C-index), receiver operating characteristic (ROC) curve, decision curve analysis (DCA) and clinical impact curve (CIC). RESULTS: A total of 343 TD cases were included in the final analysis, of which 30.32% had behavioral problems. The prediction model showed age 12-16 years, abnormal birth history, parenting pattern of indulgence, parent/close relatives with psychiatric disorders, chronic motor or vocal tic disorder (CTD)/Tourette syndrome (TS) and moderate/severe tic severity were associated with behavioral problems in children with TD. The C-index of the prediction model (nomogram) was 0.763 (95% confidence interval, 0.710 ~ 0.816). The nomogram was feasible for making beneficial clinical decisions, according to the satisfactory results of the DCA and CIC. CONCLUSIONS: A nomogram prediction model for comorbid behavioral problems in children with TD was established. The prediction model demonstrated a good discriminative ability and predictive performance for beneficial clinical decisions. This model further provides a comprehensive understanding of associated sociodemographic and clinical characteristics by visual graphs and allows clinicians to rapidly identify patients with a higher risk of behavioral problems and tailor necessary interventions to improve clinical outcomes.

Exploring Social Support in an Online Support Community for Tourette Syndrome and Tic Disorders: Analysis of Postings.

BACKGROUND: Online support communities have become an accessible way of gaining social, emotional, and informational support from peers and may be particularly useful for individuals with chronic conditions. To date, there have been few studies exploring the online support available for tic disorders, such as Tourette syndrome. An exploratory study looking at users' experiences with using online support communities for tic disorders suggested that members used such communities to share experiences, information, and strategies for tic management. OBJECTIVE: To build on these preliminary findings, this study examined the provision of social support in an online community for Tourette syndrome. METHODS: Data were collected from one publicly available online support community for Tourette syndrome and tics, from its inception to December 2019, by randomly selecting 10% of posts and their corresponding comments from each year for analysis. This resulted in 510 unique posts and 3802 comments posted from 1270 unique usernames. The data were analyzed using inductive thematic analysis. RESULTS: The findings of this study suggest that users utilized the online community as a multifaceted virtual place where they could share and ask for information about tics, unload and share their feelings arising from living with Tourette syndrome, find people facing similar situations and experiences, and freely share the realities of living with Tourette syndrome. CONCLUSIONS: The results complement the findings from a preliminary study and suggest that online support communities have a potentially valuable role as a mechanism for sharing and gaining information on illness experiences from similar peers experiencing tics and can promote self-management of tics. Limitations and recommendations for future research are discussed.

Dopamine-2 receptor antibody encephalitis presenting as pure tongue-biting in a tourette syndrome patient: a case report.

BACKGROUND: Tourette syndrome (TS) is a neuropsychiatric disorder characterized by repetitive and patterned tics. Its onset correlates with dysfunctions in immunological activation and neurotransmitters. Autoimmune movement disorders such as dopamine-2 receptor antibody encephalitis (D2R encephalitis) may go undiagnosed in TS patients seeking medical help for tic symptoms only. Here, we present a clinical case of D2R encephalitis in a TS patient. CASE PRESENTATION: A 13-year-old boy with a history of TS presented with acute tongue-biting without positive neurologic examination or auxiliary examination results, except for a weakly positive finding for D2R antibodies in the serum sample. He was initially diagnosed with possible D2R encephalitis, but the influence of TS could not be ruled out. In addition to psychotropics, we administered immunotherapy early based on clinical characteristics, and his symptoms were ameliorated significantly. During the follow-up, he was diagnosed with definite D2R encephalitis, and the dosage of psychotropics was further adjusted for fluctuating symptoms. CONCLUSIONS: Our case suggests that clinicians should discern D2R encephalitis in TS patients when tics are the primary symptoms. Administering immunotherapy early, according to clinical characteristics, may benefit the patient. Moreover, the features of premonitory urges could help evaluate the state of TS.

Living with tics: Nursing care of pediatric tourette syndrome.

Tourette syndrome (TS) is a neurodevelopmental movement characterized by abrupt, rapid, repetitive, and non-rhythmic tics. While TS is not life-threatening, such tics and comorbidities affect a children's physical and mental health and social interactions. Treatment is necessary when the children experience physical discomfort, functional dysfunction, and poor interpersonal interaction and mental health. Common management strategies for TS include psychological interventions and pharmacological treatment. Additionally, it is important to consider the individual needs of youths with TS and reassess the treatment outcomes and the need to receive continuous treatment regularly. This review summarizes the symptom management and nursing care measures for youths with TS. Understanding and listening to the healthcare problems of youths with TS can help providers develop healthcare interventions tailored to the youths' needs. Providing the youths with strategies to reduce the tics' interference with their learning outcomes and helping them develop strategies to cope with diverse interpersonal interactions at school are also important. Further, enhancing acceptance given by parents, teachers, and peers, as well as improving the general public's understanding of TS are important measures that promote the youths' social adjustment as well as their ability to coexist with TS.

Assessing response, remission, and treatment resistance in patients with obsessive-compulsive disorder with and without tic disorders: results from a multicenter study.

BACKGROUND: Highlighting the relationship between obsessive-compulsive disorder (OCD) and tic disorder (TD), two highly disabling, comorbid, and difficult-to-treat conditions, Diagnostic and Statistical Manual of Mental Disorders, fifth edition (DSM-5) acknowledged a new "tic-related" specifier for OCD, ie, obsessive-compulsive tic-related disorder (OCTD). As patients with OCTD may frequently show poor treatment response, the aim of this multicenter study was to investigate rates and clinical correlates of response, remission, and treatment resistance in a large multicenter sample of OCD patients with versus without tics. METHODS: A sample of 398 patients with a DSM-5 diagnosis of OCD with and without comorbid TD was assessed from 10 different psychiatric departments across Italy. For the purpose of the study, treatment response profiles in the whole sample were analyzed comparing the rates of response, remission, and treatment-resistance as well as related clinical features. Multivariate logistic regressions were performed to identify possible factors associated with treatment response. RESULTS: The remission group was associated with later ages of onset of TD and OCD. Moreover, significantly higher rates of psychiatric comorbidities, TD, and lifetime suicidal ideation and attempts emerged in the treatment-resistant group, with larger degrees of perceived worsened quality of life and family involvement. CONCLUSIONS: Although remission was associated with later ages of OCD and TD onset, specific clinical factors, such as early onset and presence of psychiatric comorbidities and concomitant TD, predicted a worse treatment response with a significant impairment in quality of life for both patients and their caregivers, suggesting a worse profile of treatment response for patients with OCTD.

Potential impact of COVID-19 on ongoing clinical trials: a simulation study with the neurological Yale Global Tic Severity Scale based on the CANNA-TICS study.

The COVID-19 pandemic has manifold impacts on clinical trials. In response, drug regulatory agencies and public health bodies have issued guidance on how to assess potential impacts on ongoing clinical trials and stress the importance of a risk-assessment as a pre-requisite for modifications to the clinical trial conduct. This article presents a simulation study to assess the impact on the power of an ongoing clinical trial without the need to unblind trial data and compromise trial integrity. In the context of the CANNA-TICS trial, investigating the effect of nabiximols on reducing the total tic score of the Yale Global Tic Severity Scale (YGTSS-TTS) in patients with chronic tic disorders and Tourette syndrome, the impact of the two COVID-19 related intercurrent events handled by a treatment policy strategy is investigated using a multiplicative and additive data generating model. The empirical power is examined for the analysis of the YGTSS-TTS as a continuous and dichotomized endpoint using analysis techniques adjusted and unadjusted for the occurrence of the intercurrent event. In the investigated scenarios, the simulation studies showed that substantial power losses are possible, potentially making sample size increases necessary to retain sufficient power. However, we were also able to identify scenarios with only limited loss of power. By adjusting for the occurrence of the intercurrent event, the power loss could be diminished to different degrees in most scenarios. In summary, the presented risk assessment approach may support decisions on trial modifications like sample size increases, while maintaining trial integrity.

Suicidal ideation and suicidal attempts in patients with obsessive-compulsive tic-related disorder vs obsessive-compulsive disorder: results of a multicenter Italian study.

BACKGROUND: Obsessive-compulsive disorder (OCD) and tic disorder (TD) represent highly disabling, chronic and often comorbid psychiatric conditions. While recent studies showed a high risk of suicide for patients with OCD, little is known about those patients with comorbid TD (OCTD). Aim of this study was to characterize suicidal behaviors among patients with OCD and OCTD. METHODS: Three hundred and thirteen outpatients with OCD (n = 157) and OCTD (n = 156) were recruited from nine different psychiatric Italian departments and assessed using an ad-hoc developed questionnaire investigating, among other domains, suicide attempt (SA) and ideation (SI). The sample was divided into four subgroups: OCD with SA (OCD-SA), OCD without SA (OCD-noSA), OCTD with SA (OCTD-SA), and OCTD without SA (OCTD-noSA). RESULTS: No differences between groups were found in terms of SI, while SA rates were significantly higher in patients with OCTD compared to patients with OCD. OCTD-SA group showed a significant male prevalence and higher unemployment rates compared to OCD-SA and OCD-noSA sample. Both OCTD-groups showed an earlier age of psychiatric comorbidity onset (other than TD) compared to the OCD-SA sample. Moreover, patients with OCTD-SA showed higher rates of other psychiatric comorbidities and positive psychiatric family history compared to the OCD-SA group and to the OCD-noSA groups. OCTD-SA and OCD-SA samples showed higher rates of antipsychotics therapies and treatment resistance compared to OCD-noSA groups. CONCLUSIONS: Patients with OCTD vs with OCD showed a significantly higher rate of SA with no differences in SI. In particular, OCTD-SA group showed different unfavorable epidemiological and clinical features which need to be confirmed in future prospective studies.

Current Management of Tics and Tourette Syndrome: Behavioral, Pharmacologic, and Surgical Treatments.

Tourette syndrome is a heterogeneous neurobehavioral disorder manifested by childhood-onset motor and phonic tics, often accompanied by a variety of behavioral comorbidities, including attention deficit and obsessive compulsive disorder. Treatment must be tailored to the needs and goals of the individual patients and their families. All patients should receive education on the condition and, if possible, engage behavioral therapy targeted towards tics and/or comorbidities. Pharmacological therapies, such as alpha agonists, topiramate, and vesicular monoamine transport type 2 inhibitors, are generally used as first-line therapies in patients with troublesome tics that are not controlled by behavioral therapy or when the latter is not available or accessible. Botulinum toxin injections can be used in patients with bothersome focal tics. Second-line therapy includes antipsychotics, such as fluphenazine, aripiprazole, risperidone, and ziprasidone. These medications are generally efficacious but carry the risk of metabolic syndrome, tardive dyskinesia, and other side effects. Much more research is needed before novel therapies such as cannabis-derived products or transcranial magnetic stimulation can be recommended. There is promise in ongoing clinical trials with D1 receptor antagonist ecopipam and other experimental therapeutics. Patients with tics that are refractory to conventional treatments may be candidates for deep brain stimulation, but further studies are needed to determine the optimal target selection.

Sensory hypersensitivity in Tourette syndrome: A review.

Tourette syndrome (TS) is a neurodevelopmental disorder defined by tics, but most patients also experience bothersome sensory phenomena, in the form of premonitory urges and/or sensory hypersensitivity. Whereas premonitory urges are temporally paired with tics, sensory hypersensitivity is a constant, heightened awareness of external and/or internal stimuli. The intensity of sensory hypersensitivity does not strongly correlate with the severity of tics or premonitory urges, suggesting it is a dissociable clinical phenomenon. At least 80% of TS patients report subjectively enhanced perception of various sensory stimuli. These same patients demonstrate normal static detection thresholds. However, individuals with TS habituate abnormally to repetitive stimuli, indicating incapacity to appropriately filter redundant sensory input, i.e. impaired sensory gating. Physiologic support for this hypothesis is provided by abnormal pre-pulse inhibition (PPI) and event-related potential (ERP) investigations. Preclinical data implicates parvalbumin-positive (PV+) interneuron dysfunction in altered sensory gating in TS and other neurodevelopment disorders. Studies probing TS sensory hypersensitivity must methodically account for comorbid psychiatric conditions, namely obsessive compulsive disorder (OCD), attention deficit hyperactivity disorder (ADHD), and autism spectrum disorder (ASD), as these entities appear to involve pathophysiologic processes shared with TS. The presence of psychiatric comorbidities in TS is associated with even more profound sensory processing dysfunction. A deepened understanding of TS sensory hypersensitivity will afford novel insights into disease mechanisms, clinical phenotype, and therapeutic management.

Depression in Tourette Syndrome. / Depresja w przebiegu zespolu Tourette'a.

OBJECTIVES: The aim of the study is to show relationship between severity of depressive symptoms and the severity of tics, declared feeling of stigmatization and feelings about the body. METHODS: The study included 13 people with Tourette syndrome and 13 people in a comparative group - matching method, taking into account compatibility of gender, age, number of years of education, and size of the place of residence. The study used: Polish adaptation of the Yale Global Tic Severity Scale (YGTSS), Questionnaire for Measuring Depression (Kwestionariusz do Pomiaru Depresji -KPD), Perceived Stigmatization Questionnaire (PSQ), Questionnaire of feelings about the body. RESULTS: Wilcoxon signed-rank test analysis showed that the declared general depression rate is significantly higher in the group of people with Tourette syndrome (Z = - 2.691; p < 0.01). The indicator differentiating the declared feeling of stigmatization among people with TS was the feeling of embarrassment due to other people's stare/the stare of bystanders (ZG) (Z = - 1.888; p < 0.05). The general assessment of one's body image is not different in the group of people with TS and comparative group, but two important factors have been distinguished: "My body is alien to me" (Z = - 1.897; p < 0.05), "It is difficult for me to understand changes in my body" (Z = 1.950; p < 0.05). CONCLUSIONS: It is concluded that the severity of tics, the feeling of stigmatization and selected body image indexes are related to both the general severity of symptoms of depression and its individual indicators.

Current pharmacotherapy for tic disorders.

Introduction: Though many unanswered questions about the pathophysiology of Tourette Syndrome remain, several pharmacotherapies for tics have been studied, with varying results in terms of efficacy and the strength of evidence.Areas covered: This literature review encompasses pharmacotherapies for tics. The pharmacotherapies discussed in this review include: alpha agonists, antipsychotics, topiramate, botulinum toxin, and dopamine depleters.Expert opinion: Once the presence of tics is confirmed and psychoeducation and support are provided to patients and caregivers, one must examine the degree of tic-related impairment and the presence of psychiatric comorbidities. These factors influence treatment decisions as the presence of comorbidity and related impairment may shift the treatment target. When selecting a medication for tics, the presence of ADHD (the most frequent comorbidity) strengthens the case for choosing an alpha agonist. The case for antipsychotic medications is strongest when tic-related impairment is severe and/or the tics are refractory to more conservative measures. All medications require drug safety monitoring procedures and reevaluation over time.

[Tourette syndrome: Research challenges to improve clinical practice]. / Syndrome de Gilles de la Tourette : défis de la recherche pour améliorer la pratique clinique.

Tourette syndrome is a neurodevelopmental disorder which is characterized by the presence of motor and phonic tics. These tics are generally more prevalent in childhood. Tics typically reach their maximum severity before puberty, around age 10 to 12. In most patients, tic severity usually decreases during late adolescence and adulthood. However, this is not true for all individuals. To date, the developmental trajectory leading to the persistence of tics into adulthood is still poorly understood. There are very few markers that can predict the evolution of tic symptoms from childhood to adulthood. Yet, while we cannot cure Tourette syndrome, it is possible to reduce tic severity with various treatments. The most common treatments are pharmacotherapy and behavioral and cognitive-behavioral therapy. However, there appears to be a limit to the proportion of tics that can be treated, since most treatments offer an average reduction in tics of no more than 50%. Thus, at first, this article reviews recent advances in treatment and symptom progression. Next, we propose some lines of research to improve the management and treatment of people with Tourette syndrome.

Neurological Soft Signs and Clinical Features of Tic-Related Obsessive-Compulsive Disorder Indicate a Unique Subtype.

Tic-related obsessive-compulsive disorder (OCD) may be a unique OCD subtype. This study examined whether neurological soft signs (NSSs) of patients with tic-related and tic-free OCD enable discrimination of these subgroups. We used the Neurological Evaluation Scale to assess 32 patients with tic-related and 94 with tic-free OCD, as well as 84 controls. Most patients with tic-related OCD were male, with earlier illness onset and poorer insight scores than those of patients with tic-free OCD. Patients with tic-related OCD had poorer motor coordination, sensory integration, and motor sequencing than did tic-free patients. Logistic regression using NSS subscale scores predicted tic-related OCD. Patients with tic-related OCD displayed greater neurodevelopmental abnormalities than did tic-free patients. NSSs of the former group suggest the need to separate this subgroup. Our results also support the newly introduced tic-related specifier in the fifth edition of the Diagnostic and statistical manual of mental disorders.

Help or hurt? How attention modulates tics under different conditions.

Tourette syndrome is a neuropsychiatric developmental disorder, characterized by tics that are often preceded by an increasingly uncomfortable urge to move. Tic frequency can increase when patients pay attention to their tics, if tics are not suppressed. This study investigates how attentions modulates urge intensity, tic frequency and arousal during free ticcing and tic suppression. Tic frequency (video recording), urge intensity (rating scale) and pupil width (pupillometry as a measure of arousal) were assessed in 23 patients with Tourette syndrome (mean age 33.48 ± 12.37; 14 male) during five attention conditions: 1) baseline, 2) watching own tics in a live video-feedback, 3) watching own tics in a previously recorded video, 4) thinking about situations that can trigger tics and 5) thinking about specific, non-tic related stimuli (distraction condition) during: a) free ticcing and b) tic suppression tic states. Urge intensity and tic frequency increased in the free ticcing condition when patients viewed their own tics live and when they thought about tic-triggering situations. In the tic suppression condition, tic frequency increased when patients watched a video of their tics, thought about their tics or were distracted. Pupil width increased significantly during the live feedback and the video condition compared to baseline in both tic states. Paying attention to own tics can be detrimental when tics are not suppressed. In contrast, paying attention to other stimuli appears detrimental when tics are suppressed, as would be the case during most current behavioural therapy techniques. However, results point to high emotional arousal and patients feeling uncomfortable when seeing themselves tic. The results also suggest that urge intensity is modulated by changes in attention in the same manner as tics and may drive change in tic frequency during free ticcing.

Effects of comorbidity on Tourette's tic severity and quality of life.

OBJECTIVE: The aim of this study is to gain more insight in the differential contributions of anxiety, depression and obsessive-compulsive (OC) symptom severity to quality of life (QoL) and tic severity in adults with Tourette Disorder (TD). METHODS: Self-reported OC symptom, anxiety and depression severity measures were used to investigate their predictive value on QoL and Tic severity in adult TD patients (N = 187), using correlation, regression, and mediation analyses. RESULTS: Tic severity has no effect on QoL. Depression severity directly reduces QoL, whereas anxiety and OC symptom severity have an indirect effect on QoL, mediated by depression severity. OC symptom severity directly affects tic severity, whereas depression and anxiety severity do not have a direct effect on tic or OC severity. Finally, anxiety severity indirectly impacts tic severity, with OC symptom severity functioning as a mediator. CONCLUSION: In line with and extending previous studies, these findings indicate that OC symptom severity directly influences tic symptom severity whereas depression severity directly influences QoL in TD. Results imply that to improve QoL in TD patients, treatment should primarily focus on diminishing OC and depressive symptom severity rather than focusing on tic reduction.

The Correlation Between the Severity of Premonitory Urges and Tic Symptoms: A Meta-Analysis.

Objectives: Premonitory urges (PUs) are defined as sensory experiences of pre-tic inner tension. Evidence suggests that most patients with Tourette syndrome experience PUs, which are transiently relived by the expression of tics. However, recent studies have revealed inconsistent results regarding the correlation between the severity of PUs and the severity of tic symptoms. Methods: A meta-analysis was performed to confirm the correlation between the severity of the urge and the severity of the expression of the tic. In total, 10 studies involving 626 patients with tic disorders were included in this meta-analysis. Results: The correlation coefficient (r) was extracted from each selected study, and a pooled correlation coefficient (r) and its 95% confidence intervals (CIs) were calculated. Sensitivity and subgroup analyses were performed to identify the potential sources of heterogeneity. The pooled correlation coefficient (r) of the relationship between the severity of PUs and tic symptoms was 0.296 (95% CI: 0.215-0.376) with an I2 of 15.2% (95% CI: 0.00-56.5) based on a fixed effects model. The correlation was stronger in adults than in children (p = 0.03). Conclusion: A slight to moderate positive correlation was observed between the severity of PUs and tic symptoms. This correlation may be affected by the age of the patients. Further research should seek to elucidate the relationships among PUs, tic suppression, and tic expression to support the development of behavioral intervention therapies to reduce tic symptoms.

Practice guideline recommendations summary: Treatment of tics in people with Tourette syndrome and chronic tic disorders.

OBJECTIVE: To make recommendations on the assessment and management of tics in people with Tourette syndrome and chronic tic disorders. METHODS: A multidisciplinary panel consisting of 9 physicians, 2 psychologists, and 2 patient representatives developed practice recommendations, integrating findings from a systematic review and following an Institute of Medicine-compliant process to ensure transparency and patient engagement. Recommendations were supported by structured rationales, integrating evidence from the systematic review, related evidence, principles of care, and inferences from evidence. RESULTS: Forty-six recommendations were made regarding the assessment and management of tics in individuals with Tourette syndrome and chronic tic disorders. These include counseling recommendations on the natural history of tic disorders, psychoeducation for teachers and peers, assessment for comorbid disorders, and periodic reassessment of the need for ongoing therapy. Treatment options should be individualized, and the choice should be the result of a collaborative decision among patient, caregiver, and clinician, during which the benefits and harms of individual treatments as well as the presence of comorbid disorders are considered. Treatment options include watchful waiting, the Comprehensive Behavioral Intervention for Tics, and medication; recommendations are provided on how to offer and monitor these therapies. Recommendations on the assessment for and use of deep brain stimulation in adults with severe, treatment-refractory tics are provided as well as suggestions for future research.