The contribution of deficits in limb position sense to the motor impairments of children with cerebral palsy, as well as other neurodevelopmental disorders, is increasingly being recognized. A more complete understanding of the development of multi-joint upper limb position sense is needed and has been limited, to date, by the absence of a reliable measurement technique to produce clinically meaningful information. The KINARM Exoskeleton’s bilateral position matching task, which involves passive movement of one of the subject’s arms to one of eight positions requiring different combinations of elbow and shoulder positions and active matching by the participant’s other arm, was evaluated for interrater and retest reliability. Intraclass correlation coefficients, absolute difference, minimum detectable difference that would be considered a significant change in performance, standard error of the measure, coefficient of variation, index of reliability, limit of agreement and confidence intervals were used to determine reliability on three measures of multi-joint position sense: (1) inter-trial variability in end-point position, (2) the ratio between actual and matched position, or spatial contraction/expansion, which provides a measure of the absolute accuracy of position matching, and (3) systematic errors in matching. Interrater index of reliability was very good to excellent with values of 72% for systematic errors in matching to 93% for contraction/expansion. Interrater intraclass correlation values were fair to excellent at 0.46 for systematic errors in matching to 0.81 for contraction/expansion. Standard errors in measurement were low and ranged from 0.002 to 0.06, for inter-trial variability and contraction/expansion respectively. Similarly, minimal detectable difference values for retest reliability ranged from 0.005 for inter-trial variability to 0.161 for contraction/expansion. Retest intraclass correlation values were fair to excellent at 0.38 for systematic errors in matching to 0.82 for contraction/expansion. Moderate to strong interrater and retest reliability and high measurement precision support the use of robot-based assessment of multi-joint position sense for developmental studies and promises to be a reliable clinical and research tool in the advancement of knowledge on sensory-motor coordination difficulties in children with neurodevelopmental disorders. / Thesis (Master, Rehabilitation Science) -- Queen's University, 2011-09-29 05:43:47.255

<p>Neurodevelopmental disorders develop over time and are characterized by a wide variety of mental, behavioral, and physical phenotypes. The categorization of neurodevelopmental disorders encompasses a broad range of conditions including intellectual disability, autism spectrum disorder, attention deficit hyperactivity disorder, cerebral palsy, schizophrenia, bipolar disorder, and epilepsy, among others. Diagnostic classifications of neurodevelopmental disorders are complicated by comorbidities among these neurodevelopmental disorders, unidentified causal genes, and growing evidence of shared genetic risk factors. </p><p>We sought to identify the genetic underpinnings of a variety of neurodevelopmental disorders, with a particular emphasis on the epilepsies, by employing next&ndash;generation sequencing to thoroughly interrogate genetic variation in the human genome/exome. First, we investigated four families presenting with a seemingly identical and previously undescribed neurodevelopmental disorder characterized by congenital microcephaly, intellectual disability, progressive cerebral atrophy, and intractable seizures. These families all exhibited an apparent autosomal recessive pattern of inheritance. Second, we investigated a heterogeneous cohort of &sim;60 undiagnosed patients, the majority of whom suffered from severe neurodevelopmental disorders with a suspected genetic etiology. Third, we investigated 264 patients with epileptic encephalopathies &mdash; severe childhood epilepsy disorders &mdash; looking specifically at infantile spasms and Lennox&ndash;Gastaut syndrome. Finally, we investigated &sim;40 large multiplex epilepsy families with complex phenotypic constellations and unclear modes of inheritance. The studied neurodevelopmental disorders exhibited a range of genetic complexity, from clear Mendelian disorders to common complex disorders, resulting in varying degrees of success in the identification of clearly causal genetic variants. </p><p>In the first project, we successfully identified the disease&ndash;causing gene. We show that recessive mutations in <italic>ASNS </italic> (encoding asparagine synthetase) are responsible for this previously undescribed neurodevelopmental disorder. We also characterized the causal mutations <italic>in vitro</italic> and studied Asns&ndash;deficient mice that mimicked aspects of the patient phenotype. This work describes ASNS deficiency as a novel neurodevelopmental disorder, identifies three distinct causal mutations in the ASNS gene, and indicates that asparagine synthesis is essential for the proper development and function of the brain.</p><p>In the second project, we exome sequenced 62 undiagnosed patients and their unaffected biological parents (trios). By analyzing all identified variants that were annotated as putatively functional and observed as a novel genotype in the probands (not observed in the unaffected parents or controls), we obtained a genetic diagnosis for 32% (20/62) of these patients. Additionally, we identify strong candidate variants in 31% (13/42) of the undiagnosed cases. We also present additional analysis methods for moving beyond traditional screens, e.g., considering only securely implicated genes, or subjecting qualifying variants from any gene to two unique analysis approaches. This work adds to the growing evidence for the utility of diagnostic exome sequencing, increases patient sizes for rare neurodevelopmental disorders (enabling more detailed analyses of the phenotypic spectrum), and proposes novel analysis approaches which will likely become beneficial as the number of sequenced undiagnosed patients grows. </p><p>In the third project, we again employ a trio&ndash;based exome sequencing design to investigate the role of <italic>de novo</italic> mutations in two classical forms of epileptic encephalopathy. We find a significant excess of <italic>de novo</italic> mutations in the &sim;4,000 genes that are the most intolerant to functional genetic variation in the human population (P = 2.9 x 10<super>&ndash;3</super>, likelihood analysis). We provide clear statistical evidence for two novel genes associated with epileptic encephalopathy &mdash; <italic>GABRB3</italic> and <italic>ALG13</italic>. Together with the 15 well&ndash;established epileptic encephalopathy genes, we statistically confirm the association of an additional ten putative epileptic encephalopathy genes. We show that only &sim;12% of epileptic encephalopathy patients in our cohort are explained by <italic>de novo</italic> mutations in one of these 24 genes, highlighting the extreme locus heterogeneity of the epileptic encephalopathies. </p><p>Finally, we investigated multiplex epilepsy families to uncover novel epilepsy susceptibility factors. Candidate variants emerging from sequencing within discovery families were further assessed by cosegregation testing, variant association testing in a case&ndash;control cohort, and gene&ndash;based resequencing in a cohort of additional multiplex epilepsy families. Despite employing multiple approaches, we did not identify any clear genetic associations with epilepsy. This work has, however, identified a set of candidates that may include real risk factors for epilepsy; the most promising of these is the <italic>MYCBP2</italic> gene. This work emphasizes the extremely high locus and allelic heterogeneity of the epilepsies and demonstrates that very large sample sizes are needed to uncover novel genetic risk factors. </p><p>Collectively, this body of work has securely implicated three novel neurodevelopmental disease genes that inform the underlying pathology of these disorders. Furthermore, in the final three studies, this work has highlighted additional candidate variants and genes that may ultimately be validated as disease&ndash;causing as sample sizes increase.</p> / Dissertation

The overall aim of this thesis was to characterize single-channel aEEG/EEG, recorded during the first postnatal days in preterm infants, in relation to brain function and two-year outcome. Study I investigated if aEEG/EEG was associated with neonatal brain injury, inflammation and outcome in 16 very preterm (VPT) infants. The interburst interval (IBI) was prolonged, and aEEG amplitudes were lower in infants with brain injury, and in infants developing handicap. Cord blood TNF-α correlated with IBI. Study II investigated inter-rater agreement of visual burst detection, as compared to automated burst detection based on a non-linear energy operator (NLEO) in an EEG data set from 12 extremely preterm (EPT) and 6 VPT infants. The sensitivity of the NLEO was 64 % and 69 % (EPT and VPT infants, respectively) and the specificity 96 % and 88 %. The algorithm was then modified to further improve the accuracy. Study III investigated if arterial carbon dioxide and plasma glucose is associated with EEG continuity. In 247 sets of samples (PaCO2, plasma glucose, IBI) from 32 EPT infants there was a positive association between PaCO2 and IBI; higher PaCO2 was associated with longer IBI. Corrected for carbon dioxide, plasma glucose had a U-shaped association with IBI in infants with good outcome. Study IV investigated the predictive value of aEEG/EEG in 41 EPT and 8 VPT infants. All VPT infants had good outcome. Predictors of outcome in EPT infants included presence or absence of burst-suppression, continuous activity and cyclicity, median IBI and interburst%. Seizures were associated with neonatal brain damage but not with outcome. Improved preterm brain monitoring may in the future be used for early identification of infants at high risk of brain damage and adverse outcome, which may have implications for direction of care and for early intervention.

Working memory is involved in a variety of cognitive tasks, with working memory capacity predicting an individual's ability to process information and focus attention on taskrelated information. Subsequently, recent research has investigated whether working memory capacity can be improved through training and whether improvements generalise to other cognitive, behavioural or emotional domains. This training is typically adaptive in nature, changing in difficulty according to participant ability, and can be completed in the participant's home on a computer, giving it the potential to be an easily accessible intervention for a range of clinical populations. The first paper presents a systematic review evaluating the effectiveness of computerised working memory training for individuals with neurodevelopmental disorders, which are often associated with working memory difficulties. The review found that to date, working memory training has been investigated in four neurodevelopmental disorders: attention deficit/hyperactivity disorder; autism spectrum disorder; intellectual disability and specific learning disorder. The findings indicate that although training appears to produce short-term improvements in the working memory capacity, this does not reliably generalise to other cognitive processes or disorder-specific symptoms. The second paper presents a randomised controlled trial investigating the effects of working memory training for reducing worry in high-worriers. Working memory capacity limitations, and subsequent difficulties in attentional control, are believed to be central to the maintenance of worry. Participants were randomly assigned to complete 15 days of nonadaptive working memory training using a 1-back task, or adaptive working memory training using a n-back task. Training led to improvements in working memory capacity and worry symptoms post-training and at four-week follow-up, with improvements on the adaptive training task significantly correlating with improvements in working memory capacity and worry. These findings are discussed in terms of implications for clinical practice and future research, together with the limitations of the study.

<b>Background:</b> The survival rate of preterm children has risen steadily due to advances in obstetric and neonatal intensive care. Children born extremely preterm (&le;28 weeks of gestation) are at high risk of long term developmental problems, including cerebral palsy, motor and cognitive impairment, visual and auditory deficits and behavioural problems. This can have serious implications for their quality of life and that of their family and carers. These children take up a disproportionate amount of neonatal intensive care unit resources and overall costs, and as they grow up are more likely to require additional health and social care services beyond routine care to compensate for their functional limitations. The early identification and management of factors that mediate long term outcome is necessary to assist healthcare professionals in selecting appropriate treatment pathways, and to develop, target and evaluate interventions. Many risk factor analyses for neurodevelopmental impairment have been published in preterm populations, but this vast literature has not been formally summarised. Furthermore, there is a dearth of studies reporting longitudinal analysis of neurodevelopmental trajectories from early childhood to adulthood. <b>Objectives:</b> The first aim of this thesis was to perform a comprehensive systematic review of the world literature over the last two decades, to consolidate the evidence about the prognosis of neurodevelopmental outcome in children born very preterm or with very low birth weight. The second aim was to conduct a longitudinal analysis of a cohort of extremely preterm participants followed up into early adulthood to investigate the trajectories of long term sequelae over time, and to examine the association of neurodevelopmental course in relation to the predictive factors identified in the systematic review. <b>Methods:</b> A systematic review was conducted using MEDLINE, EMBASE and PyscINFO databases to identify studies published between January 1 1990 and June 1 2014 reporting multivariable prediction models for the neurodevelopment of children born &le;32 weeks of gestation or with a birth weight &le;1250 grams (protocol registration number CRD42014006943). Seventy-eight studies reporting 222 risk factor models for neurodevelopmental outcome were identified. Two independent reviewers extracted key information about study design, outcome definition, risk factor selection, model development, reporting, and conducted a risk of bias assessment. To address the second objective of the study, a longitudinal analysis of cognitive and behavioural trajectories was conducted using a prospective, population-based cohort study in the United Kingdom and the Republic of Ireland. Three hundred and fifteen surviving infants born less than 26 completed weeks of gestation recruited at birth in 1995 and 160 term-born classroom peers recruited at age six were followed-up to 19 years. Participants were invited for up to four standardized, blinded cognitive assessments and the parent-completed Strengths and Difficulties Questionnaire was used to assess behavioural problems. <b>Results:</b> The systematic review of risk factors for motor impairment in children born very preterm or with very low birth weight provided strong evidence that neonatal brain injury is a robust prognostic factor for cerebral palsy, and some evidence that the use of postnatal steroids increases the risk and the use of antenatal steroids reduces the risk of cerebral palsy. There was moderate evidence that male sex was prognostic for motor impairment at school age in children free of major disability. The systematic review of risk factors for cognitive impairment identified male sex, non-white ethnicity, lower levels of parental education and lower birth weight as significant predictors of global cognitive dysfunction in early infancy, with parental education having a sustained impact after five years of age. There was also evidence that male sex was predictive of delayed language development in early infancy. Gestational age was found to be of limited use as prognostic factor for cerebral palsy, motor and cognitive impairment in cohorts restricted to &le;32 weeks of gestation. There was a dearth of good quality studies investigating risk factors for behavioural problems and psychiatric disorders and the findings of this review were inconclusive. The only factors that appeared to be consistent predictors of general behavioural problems were markers of socio-economic deprivation, neurodevelopmental or cognitive delay, and an abnormal behavioural screen in early infancy. In the longitudinal analysis of the prospective, population-based cohort of extremely preterm children, cognitive trajectories were stable in both the extremely preterm and term-born groups over time with persistent deficit in the extremely preterm group of 25.2 IQ points (95&percnt; CI: -27.8 to -22.6, p&LT;0.001) and only minimal catch-up over time. Participants with neonatal brain injury and of male sex had the largest deficits, but a lower level of maternal education and earlier gestational age at birth were also associated with reduced IQ scores. Behavioural problems were also more prevalent among the extremely preterm participants who had a mean Total Difficulties Score of 4.81 points above their term-born peers (95&percnt; CI: 3.76 to 5.87, p&LT;0.001) and which persisted over the time period. Behavioural difficulties were mainly due to hyperactivity, inattention and peer problems and were strongly associated with a positive behavioural screen in early infancy. <b>Conclusions:</b> The most robust predictors of poor neurodevelopmental outcome identified by the systematic review were neonatal brain injury, male sex, and markers of social disadvantage. The unclear findings for many risk factors may reflect differences in study design, study population, methodological quality and lack of standardization of measures. Or it may simply reflect the fact that prognostic modelling in such a heterogeneous population is challenging and complex, with multiple risk factors acting sequentially over time, and often with the existence of multiple impairments within the same individual. The main conclusions from the longitudinal analysis of children born extremely preterm is that being born too soon appears to place limits on brain plasticity and function which is not recovered over time; with the most vulnerable being males and those with evidence of brain injury early in life. These structural abnormalities may disturb neurodevelopmental processes and impede the brain from maintaining a normal developmental trajectory. If extremely preterm children fail to achieve optimum levels of cognitive function and are still experiencing behavioural problems once they have reached maturity, then this has implications for health and well-being in later adulthood and old age. Cognitive test scores in infancy and early childhood reflect early adult outcomes and a positive behavioural screen in infancy is strongly associated with early adult behavioural outcomes. <b>Recommendations:</b> The systematic review revealed some shortcomings in methodology and reporting that could be improved in future studies, and confirmed that that there is a dearth of properly designed and well-conducted prognostic modelling studies in this field. The findings and recommendations of this critical review should be used as a basis for the design, analysis and reporting of future studies seeking to develop multivariate risk factor or prognostic models in this population. There is an urgent need for larger population cohorts followed up routinely beyond two years as subtle outcomes such as impairment of executive function and fine motor skills cannot be reliably assessed at this age, and the natural course of some disorders may have their onset later in childhood. / Studies with larger sample sizes and greater power are needed for studying less common conditions in preterm populations and there should be more standardisation of outcome and risk factor measurements, particularly with the use of standard diagnostic evaluations to assess psychiatric disorders. Future studies should include a term-born comparison group and adopt appropriate statistical analysis techniques to analyse longitudinal outcome data and the impact of risk factors on these trajectories. Additional research is required to improve the prediction of individual differences, and to identify the neuropathological differences underlying different developmental trajectories and their interaction with environmental influences over time.

Young adolescents with neurodevelopmental disabilities tend to have lower levels of participation and poorer well-being compare to their typically developing peers. School is a key setting for adolescents to participate. School’s social environment is found to be influential to both participation and well-being for young adolescents. This paper investigates the relations between school’s social environment and participation, well-being of young adolescents with self-rated neurodevelopmental problems (SNP) and whether participation is associated with well-being of this population. Data of this study was retrieved from wave three of the longitudinal research programme LoRDIA. Study participants were 175 adolescents aged 14-15 years old with SNP. Linear regression was conducted to test the hypothesis. Results show that bullying is the strongest (negative) predictor to both participation and well-being for young adolescents with SNP, which means students within this group who have been bullied are more likely to have low level participation and poor well-being. Relation to teachers is also a strong positive predictor to participation of this population. Class atmosphere is less predictive compare to the other two predictors but still have moderate positive correlation with both participation and well-being. Adolescents with neurodevelopmental disabilities or problems are a vulnerable group, school’s environment is essential to their development. Experience of bullying has a profound and long-term negative effect on children’s participation and well-being. Therefore, more longitudinal data will be needed to further understand this issue. Relation to teachers brings up that the focus and attention should not only been put on students but also on fulfilling teacher’s needs, so they could provide higher quality of teacher-student relationships.

Compreender as bases do comportamento social e do desenvolvimento socioafetivo humano é essencial tanto para indivíduos com desenvolvimento típico (DT) quanto com transtornos neuropsiquiátricos. A Síndrome de Williams (SW) é uma condição neurogenética rara ocasionada pela deleção de aproximadamente 28 genes no cromossomo 7q11.23. A sintomatologia inclui desde dismorfismos faciais a alterações do funcionamento cognitivo e socioafetivo, com a presença de deficiência intelectual de grau leve a moderado. O processamento de estímulos afetivos tem sido foco de grande interesse em indivíduos com SW. Apesar de parte das pesquisas apontarem que esta população tem habilidade preservada de reconhecimento de expressões facias de emoções positivas e prejuízos no reconhecimento de emoções negativas, este ainda não é um campo consensual. Ao mesmo tempo, estudos indicam maior interesse desta população em relação a música e diferenças no neuroprocessamento de trechos musicais com valência afetiva. O presente trabalho teve por objetivo caracterizar o processamento de estímulos afetivos musicais e visuais em crianças e adolescentes com Síndrome de Williams. O Estudo I buscou validar trechos musicais com valência afetiva em cultura brasileira e analisar o efeito do treino musical na compreensão de emoções em música. Músicas com valência afetiva foram avaliadas pelos participantes de maneira correspondente à emoção pretendida pelo compositor e de forma similar entre as populações brasileiras e canadenses. O efeito do treino musical sobre a habilidade de reconhecer as emoções em música tiveram maior impacto em emoções com maior grau de dificuldade para os participantes como todo. O Estudo II visou caracterizar o perfil musical de crianças e adolescentes com SW e diferenciar o processamento de estímulos afetivos musicais em crianças e adolescentes com SW com as de DT. Pessoas com SW foram avaliadas com maior habilidade musical global. Não foram encontradas diferenças no que diz respeito ao interesse por atividades musicais. O Estudo III teve como objetivos diferenciar habilidade de reconhecimento de emoções o padrão de rastreamento do olhar frente a estímulos afetivos visuais em crianças e adolescentes com SW e SW com sintomas de TEA (SW/TEA). Pessoas com SW desprenderam maior tempo de fixação nos olhos e em faces alegres quando comparadas a faces tristes. Resultados indicam diferença no reconhecimento de emoções e rastreamento de olhar em indivíduos com SW/TEA. Padrão de reconhecimento em estímulos musicais e visuais foi semelhante na população SW, com acentuado prejuízo no reconhecimento de emoções negativas e preservação do reconhecimento de emoções positivas. Este achado reforça a modularidade do processamento neurológico das emoções básicas. Crianças com SW reconheceram mais facilmente estímulos musicais de valência positiva em comparação aos visuais sugerindo que o domínio da música seja um ponto de força desta população / Understand the foundation of social behavior and human social and affective development is essential for both individuals with typical developmental (TD) and neuropsychiatric disorders. Williams Syndrome (WS) is a rare neurogenetic condition caused by the deletion of approximately 28 genes on chromosome 7q11.23. The symptomatology includes from facial dysmorphisms to changes in cognitive and social and affective functioning, with the presence of mild to moderate intellectual deficiency. The processing of affective stimuli has been a focus of great interest in individuals with WS. Although part of the research indicates that this population has preserved ability to recognize face expressions of positive emotions and impairment in the recognition of negative emotions, this is not yet a consensual field. At the same time, studies indicate greater interest of this population in relation to music and differences in the neuroprocessing of musical excerpts with affective valence. The present work aimed to characterize the processing of musical and visual affective stimuli in children and adolescents with Williams Syndrome. Study I sought to validate musical excerpts with affective valence in Brazilian culture and to analyze the effect of musical training on the understanding of emotions in music. Songs with affective valence were evaluated by the participants corresponding to the emotion pretended by the composer and similarly between the Brazilian and Canadian populations. The effect of musical training on the ability to recognize emotions in music has had a greater impact on emotions with a greater degree of difficulty for participants as a whole. Study II aimed to characterize the musical profile of children and adolescents with WS and to differentiate the processing of musical affective stimuli in children and adolescents with WS compered to TD. People with WS were assessed with greater overall musical ability. No differences were found regarding the interest in musical activities. The aim of Study III was to differentiate between the ability to recognize emotions and the pattern of eye tracking in relation to visual affective stimuli in children and adolescents with SW and WS with ASD symptoms. People with SW gave more fixation time to the eyes and happy faces when compared to sad faces. Results indicate difference in the recognition of emotions and eye tracking in individuals with SW / ASD. Recognition pattern in musical and visual stimuli was similar in the WS population, with marked impairment in the recognition of negative emotions and preservation of the recognition of positive emotions. This finding reinforces the modularity of neurological processing of basic emotions. Children with WS recognized easily positive musical stimuli compared to visual ones suggesting that the domain of music is the strength of this population

This thesis, which focuses on sleep disturbance in people with neurodevelopmental disabilities, is divided into three sections. Paper one is a systematic review of the extant literature on objective studies of sleep in neurodevelopmental genetic disorders. Twenty papers met inclusion criteria and were subject to quality assessment, of which five were found to be high-quality, thirteen were medium-quality and two were low-quality. Studies were grouped by disorder and although there was some disparity across investigations, generally there was agreement about specific sleep difficulties in each disorder which seem to be part of the behavioural phenotypes. Overall a lack of total sleep, diminished REM sleep, and fragmented, less efficient sleep are prevalent across the disorders. Paper two is an empirical study which employed actigraphy to assess sleep in children with mucopolysaccharidosis type III (MPS III) and typically developing children. Parents completed a sleep diary, a sleep questionnaire and took saliva samples from their child. Actigraphic findings showed that MPS III patients had lengthened sleep onset latencies and greater daytime sleep than controls, but night-time sleep duration was within the normal range. In the MPS III group, some sleep problems correlated with age and progression of the disorder. Analysis of saliva samples revealed that children with MPS III had abnormal melatonin concentrations. Questionnaire responses demonstrated that children with MPS III had more sleep difficulties in all domains compared to controls. Implications for the management of sleep difficulties are discussed. Paper three is a critical appraisal of the research process which includes personal reflections on designing and conducting this research and a discussion of the principal issues which arose. Strengths and limitations of the research, ideas for further research and implications for clinical practice are considered.

Tic disorders can have a significant emotional and social impact on children and their families. There is increasing support for the use of parenting programmes in childhood conditions; however research into the applicability of such programmes in tic disorders is limited. This thesis therefore aimed to investigate the topic of parenting interventions in tic disorders. The thesis presents five chapters, written as a series of self-contained papers and prepared in accordance with selected journal submission guidance. Paper 1 is a systematic literature review of the implementation and effectiveness of behavioural parent training programmes across neurodevelopmental disorders. Twenty-two randomised controlled studies were included in the review. Neurodevelopmental disorders included attention deficit hyperactivity disorder, autistic spectrum conditions, intellectual difficulties and developmental disorders and tic disorders. Training programmes included Triple P, Barkley’s Defiant Children, Parent Child Interaction Therapy, New Forest Parenting, Parenting your Hyperactive Preschooler, Parents Plus Children, Preschoolers with Autism, Incredible Years and an idiosyncratic programme. Effects of interventions on child outcomes were examined alongside intervention characteristics and content. Results indicated robust evidence for effects of parent training on attention-deficit/hyperactivity difficulties and externalising difficulties, across neurodevelopmental disorders and training programmes. Evidence to support the effect of parent training on internalising, social and global difficulties across disorders was less robust, with fewer and inconsistent outcomes reported. Clinical and research implications resulting from the findings are discussed. Papers 2 and 3 present empirical studies. Both studies are Q-methodological investigations into opinions on parenting interventions in tic disorders. Q-methodology is a technique which enables participants’ subjective viewpoints to be grouped using by-person factor analysis. Views on the acceptability, effectiveness, feasibility and utility of parenting interventions were explored across parents of children with tic disorders (Paper 2) and professionals (Paper 3). Across both studies, seven main factors were identified (parents four, professionals three). Findings highlighted that interventions were generally considered acceptable, justified and perceived as needed. However, important differences in opinions were found within and between parent and professional groups, highlighting key clinical considerations for possible intervention format, delivery and content. Paper 4 provides a commentary of the clinical implications of these findings when the two studies are considered together, and provides guidance to further develop and implement interventions. Paper 5 presents a critical review, including discussion of the strengths, limitations and implications of the findings, alongside personal reflections on the research process.

Participants will be able to identify specific nutritional concerns with neurodevlopmental diagnoses and related feeding challenges in children, discuss the importance of interprofessional approaches to care, and the role of the RD in treatment of this unique population.