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. WHAT THIS PAPER ADDS: Three measures provided studies on content validity and psychometric measurement properties. The Participation and Sensory Environment Questionnaire-Home had moderate quality for content validity studies and high-to-moderate quality evidence for psychometric properties. The Participation and Sensory Environment Questionnaire was the only measure that included consumer involvement through qualitative interviews and pilot testing. Consumer involvement in measure development is important for content validity. Ongoing evaluation of measures against contemporaneous standards is recommended.

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.

Sensory dysregulation in tic disorders is associated with executive dysfunction and comorbidities.

BACKGROUND: Tics are conceptualized as a sensorimotor phenomenon with a premonitory urge typically described by patients. As observed in other neurodevelopmental disorders, we have observed sensory dysregulation symptoms, such as tactile hypersensitivity to clothing, in children with tic disorders; however, formal clinical research in this area is limited. OBJECTIVE: To define the presence of sensory dysregulation symptoms in tic disorders, and their clinical associations. METHODS: Prevalence of sensory dysregulation in 102 children with tic disorders was compared to 61 age- and sex-matched healthy controls. Sensory dysregulation, executive function, and quality of life data were obtained through the Short Sensory Profile-2, Sensory Profile-2, Sensory Processing Measure, Behaviour Rating Inventory of Executive Function-2, and Strength and Difficulties Questionnaire and Pediatric Quality of Life Inventory. Tics were assessed with the Yale Global Tic Severity Scale. RESULTS: Children with tics, in the presence of comorbidity, had elevated sensory dysregulation compared to healthy controls (P < 0.001). There was a positive correlation between sensory dysregulation and global executive difficulties in children with tics and comorbidity (n = 87; rho = 0.716; P < 0.001) and a negative correlation of sensory dysregulation with quality of life (n = 87; rho = -0.595; P < 0.001). In children with tics, there was an association between sensory dysregulation and number of comorbidities (P < 0.001). CONCLUSION: In the presence of comorbidity, children with tic disorders have broad sensory dysregulation symptoms beyond the premonitory urge. There was a statistically significant association between sensory dysregulation and executive function difficulties and the presence of neurodevelopmental and psychiatric comorbidity. Sensory dysregulation can be considered neurodevelopmental symptoms, providing insight into the neurobiology of tics and opportunities for therapeutic intervention. © 2019 International Parkinson and Movement Disorder Society.

An exploratory study into an adapted use of the Alert Program for tic disorder in children.

OBJECTIVES: This preliminary study explored whether an adapted approach to the Alert Program, that uses sensorimotor strategies, might assist with management of tic disorders in children. The Alert Program, a program that uses sensorimotor strategies for self-regulation in children with neurodevelopmental disorders, had not been trialled with children with tic disorders. METHODS: Ten children with tic disorder were assessed using the Dunn Sensory Profile 2 (SP2), the Yale Global Tic Severity Scale (YGTSS) and the Parent Tic Questionnaire (PTQ). Participants attended three 60-90-minute appointments with an occupational therapist and clinical psychologist for implementation of the adapted Alert Program. RESULTS: The YGTSS showed tic reduction in all participants. The total YGTSS pre-intervention mean score of 46.5 improved to 17.7 post-therapy. Five participants reported no impairment post-therapy. PTQ scores reduced in nine participants. On the SP2, 30% of participants scored as having sensory sensitivities that impaired daily function. CONCLUSIONS: This exploratory study found trialling an adapted approach to the Alert Program that uses sensorimotor-based approach decreased tic severity in children with tic disorders. A randomised controlled trial is needed to establish the effectiveness and feasibility of this approach.