Background: Though Gilles de la Tourette's syndrome (GTS) has significant impact on the quality of life of its patients, measures of health-related quality of life (HR-QOL) specific to adolescents and adults with GTS were not developed until recently. The present study provides evidence on the validity of the Gilles de la Tourette Syndrome-Quality of Life Scale (GTS-QOL), the first disease-specific HR-QOL instrument for GTS patients, for the first time in an East Asian sample.Methods: One hundred and two Japanese individuals aged 13 and above with GTS were included in our study. Internal consistency was evaluated using Cronbach's alpha. The 4-factor structure of the GTS-QOL was assessed using confirmatory factor analysis, using goodness of fit indices, factor loadings of each questionnaire item, and covariances between factors. Validity was assessed using interscale correlations. Convergent and discriminate construct validity was evaluated using correlations with other scales such as the 28-item General Health Questionnaire, the Yale Global Tic Severity Scale, and the short version of the Padua Inventory.Results: Scaling assumptions were met. Internal consistency reliability was high, with a Cronbach's alpha of 0.96. Confirmatory factor analysis revealed sufficient factor loadings and goodness of fit. All measures of goodness of fit corroborated the fit of the 4-factor model. Standardized covariances between factors in the confirmatory factor analysis were >0.8. There were significant correlations with other well-validated scales, and thus convergent and discriminate construct validity was sufficient.Conclusion: The GTS-QOL is a valid and reliable instrument to measure disease-specific HR-QOL of GTS patients in Japan.
Background Many children 4 to 6 years old exhibit compulsive-like behavior, often with comorbid Tourette symptoms, making this age group critical for investigating the effects of having comorbid Tourette symptoms with compulsive-like behavior. However, these effects have not yet been elucidated: it is unclear whether having comorbid tics with compulsive-like behavior leads to lower quality of life. This cross-sectional study aims to investigate the effect of comorbid Tourette symptoms on distress caused by compulsive-like behavior in very young children. Methods Self-administered questionnaires were distributed to guardians of children aged 4 to 6 attending any of the 59 public preschools in a certain ward in Tokyo, Japan. The questionnaire contained questions on the presence of Tourette symptoms, the presence of specific motor and vocal tics, frequency/intensity of compulsive-like behavior, and the distress caused by compulsive-like behavior, which was rated on a scale of 1 to 5. Additionally, questions on autism spectrum disorder (ASD) traits, attention-deficit/hyperactivity disorder (ADHD) traits, internalizing behavior traits, and externalizing behavior traits were included in the questionnaire as possible confounders of distress caused by compulsive-like behavior. Wilcoxon rank-sum tests were conducted to compare the distress caused by compulsive-like behavior and frequency/intensity of compulsive-like behavior between children in the Tourette symptoms group and the non-Tourette symptoms group. Furthermore, a stepwise regression analysis was performed to assess the effects of the independent variables on distress caused by compulsive-like behavior. Another stepwise regression analysis was performed to assess the relationship between distress caused by compulsive-like behavior and the presence of five specific motor and vocal tics. Results Of the 675 eligible participants, distress due to compulsive-like behavior was significantly higher in children in the Tourette symptoms group compared to the non-Tourette symptoms group (2.00 vs 1.00, P < 0.001). Stepwise regression analysis showed that frequency/intensity of compulsive-like behavior, being in the Tourette symptoms group, ASD traits, and internalizing behavior traits were predictors of distress due to compulsive-like behavior. Two specific tics, repetitive noises and sounds and repetitive neck, shoulder, or trunk movements, were significant predictors of distress due to compulsive-like behavior. Conclusions Comorbid Tourette symptoms may worsen distress caused by compulsive-like behavior in children 4 to 6 years old, and specific motor and vocal tics may lead to greater distress.
Background Associations between tic disorders and other neurodevelopmental disorders, such as stuttering, dyslexia, and developmental coordination disorder (DCD) in preschool children are unknown, especially in Japan. This study investigated these associations and their relationship with the support requirements using new screening tool named “Check List of obscure disabilities in Preschoolers” (CLASP). Methods We distributed questionnaires to the guardians of children aged 5 or 6 years old in the final grade of preschool. In total, we collected 103 responses. After excluding the responses with missing answers and those that did not provide consent, we analyzed 98 responses. We used t-tests with unequal variances and Spearman’s correlation coefficients to investigate the associations between tic disorders and other neurodevelopmental disorders. We also conducted a two-step hierarchical multiple regression analysis to examine how the severity of tic and other neurodevelopmental disorders symptoms was associated with the support requirements of parents. Results T-tests and correlation analyses suggested weak relationships between tics and stuttering and dyslexia. Conversely, tics and DCD had a significant and robust correlation. Furthermore, multiple regression analyses demonstrated that tics and DCD were significantly associated with parents requiring support. Conclusions These results suggest a relationship between tics and DCD in preschool children. More research on these relationships could promote additional support for children of this age with tic and neurodevelopmental disorders and their parents.
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