A sensory approach to the assessment and treatment of children with tic disorders

Tic disorders are the most common movement disorder in childhood. Tics interfere with the child’s quality of life and functions. With 80-90% of children with tics experiencing comorbid neurodevelopmental disorders, assessing and treating children with tic disorders is complex. Broader hypersensitivities have been reported beyond the premonitory urge in children with tics. There is limited research into sensory dysregulation in patients with tics. With no available disease-modifying or curative therapies for tics, there is a clinical need for research into the prevalence of sensory dysregulation in children with tics and to explore a sensory-based approach to reduce tics. This thesis details four studies that aimed to improve the understanding of the prevalence of sensory dysregulation, assessment, and treatment of children with tic disorders. Children with tics (n=102) were compared with age- and sex-matched healthy controls (n=61). Children with tics and comorbidities have significant sensory dysregulation compared to healthy controls (P<0.001). A pilot study of a sensory-motor treatment with children with tic disorders (n=10) achieved tic reduction in all participants (Yale Goble Tic Severity Scale mean improvement score of 28.8). A systematic review was conducted to identify sensory measures with good psychometric properties to comprehensively assess sensory dysregulation with children with tics. Of the 12 proxy-report sensory-based measures from 20 included articles, no measure included all sensory dysregulation experienced by study participants. A qualitative study with 16 families with children with tics was conducted. Using thematic analysis, the findings showed a need for a new proxy-report sensory-based measure for children with tics. This research provides an extensive understanding of the prevalence of sensory dysregulation, breath of sensory experiences and the effectiveness of a sensory-motor approach to manage tics in children with tic disorders

Maternal autoimmunity and inflammation are associated with childhood tics and obsessive-compulsive disorder: Transcriptomic data show common enriched innate immune pathways.

Although genetic variation is a major risk factor of neurodevelopmental disorders, environmental factors during pregnancy and early life are also important in disease expression. Animal models demonstrate that maternal inflammation causes fetal neuroinflammation and neurodevelopmental deficits, and brain transcriptomics of neurodevelopmental disorders in humans show upregulated differentially expressed genes are enriched in immune pathways. We prospectively recruited 200 sequentially referred children with tic disorders/obsessive-compulsive disorder (OCD), 100 autoimmune neurological controls, and 100 age-matched healthy controls. A structured interview captured the maternal and family history of autoimmune disease and other pro-inflammatory states. Maternal blood and published Tourette brain transcriptomes were analysed for overlapping enriched pathways. Mothers of children with tics/OCD had a higher rate of autoimmune disease compared with mothers of children with autoimmune neurological conditions (p = 0.054), and mothers of healthy controls (p = 0.0004). Autoimmunity was similarly elevated in first- and second-degree maternal relatives of children with tics/OCD (p 0.0001 and p = 0.014 respectively). Other pro-inflammatory states were also more common in mothers of children with tics/OCD than controls (p 0.0001). Upregulated differentially expressed genes in maternal autoimmune disease and Tourette brain transcriptomes were commonly enriched in innate immune processes. Pro-inflammatory states, including autoimmune disease, are more common in the mothers and families of children with tics/OCD. Exploratory transcriptome analysis indicates innate immune signalling may link maternal inflammation and childhood tics/OCD. Targeting inflammation may represent preventative strategies in pregnancy and treatment opportunities for children with neurodevelopmental disorders

Proxy‐reported sensory measures for children and adolescents with neurodevelopmental disorders: A systematic review

AIM: To determine the quality and utility of proxy‐reported sensory measures for children and adolescents with neurodevelopmental disorders (such as autism spectrum disorder, attention‐deficit/hyperactivity disorder, movement disorders, and intellectual disability). METHOD: We systematically searched 11 databases. We applied the updated Consensus‐based Standards for the selection of health Measurement INstruments (COSMIN) Risk of Bias checklist and criteria for good measurement properties to evaluate instrument development and psychometric properties. Findings were summarized using a COSMIN adaptation of Grading of Recommendations, Assessment, Development and Evaluations. RESULTS: From 11 databases, 6748 articles were screened. Ninety‐one full‐length articles were reviewed after removing excluded studies and manual searches conducted by two reviewers. Data were extracted for 12 measures from 20 articles. Of the 12 measures, only three provided sufficient data to evaluate content validity and psychometric measurement properties. The Participation and Sensory Environment Questionnaire‐Home (PSEQ‐H) was the only measure that satisfied moderate content validity and moderate‐to‐high quality for measurement properties. These properties included: structural validity, hypothesis testing for construct validity, internal consistency, reliability, and measurement error. INTERPRETATION: One measure, the PSEQ‐H, met eight criteria for good measurement properties. To facilitate evidence‐informed clinical decision‐making, all psychometric properties of all 12 sensory‐based, proxy‐reported measures were presented. The importance of consumer engagement in measure development and the need for ongoing evaluation of measures against contemporaneous standards is recommended