We investigate the role of the mitochondrion, an organelle highly sensitive to environmental agents, in the influence of prenatal air pollution exposure on neurodevelopment and behavior in 96 children with autism spectrum disorder (ASD) [45 with neurodevelopmental regression (NDR); 76% Male; mean (SD) age 10 y 9 m (3 y 9 m)]. Mitochondrial function was assessed using the Seahorse XFe96 in fresh peripheral blood mononuclear cells. Second and third trimester average and maximal daily exposure to fine air particulate matter of diameter ≤2.5 µm (PM2.5) was obtained from the Environmental Protection Agency’s Air Quality System. Neurodevelopment was measured using the Vineland Adaptive Behavior Scale 2nd edition and behavior was assessed using the Aberrant Behavior Checklist and Social Responsiveness Scale. Prenatal PM2.5 exposure influenced mitochondrial respiration during childhood, but this relationship was different for those with (r = 0.25–0.40) and without (r = −0.07 to −0.19) NDR. Mediation analysis found that mitochondrial respiration linked to energy production accounted for 25% (SD = 2%) and 10% (SD = 2%) of the effect of average prenatal PM2.5 exposure on neurodevelopment and behavioral symptoms, respectively. Structural equation models estimated that PM2.5 and mitochondrial respiration accounted for 34% (SD = 4%) and 36% (SD = 3%) of the effect on neurodevelopment, respectively, and that behavior was indirectly influenced by mitochondrial respiration through neurodevelopment but directly influenced by prenatal PM2.5. Our results suggest that prenatal exposure to PM2.5 disrupts neurodevelopment and behavior through complex mechanisms, including long-term changes in mitochondrial respiration and that patterns of early development need to be considered when studying the influence of environmental agents on neurodevelopmental outcomes.

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Prenatal air pollution influences neurodevelopment and behavior in autism spectrum disorder by modulating mitochondrial physiology.

As adaptive skills (AS) are dynamic and may indicate the success of an intervention, they are a common domain measured in clinical trials. Typical interview tools for measuring AS are time-consuming, and questionnaire measures often lead to inconsistent information. The present study was designed to evaluate the feasibility, validity and test-retest performance of the Pediatric Evaluation of Disability Inventory Computer Adaptive Test (PEDI-CAT) in Fragile X syndrome (FXS). The PEDI-CAT is administered via tablet and uses the item response theory to efficiently determine the items administered. The PEDI-CAT was administered to 42 individuals with FXS (27 males; 15 females) aged 1.6-50.9 years (M = 14.9; SD = 11.2), followed by the Vineland-3 (VABS-3) interview for comparison. Administration was efficient (M = 21.7 min; SD = 9.5; range 8-45 min; mode = 19). Males and females did not significantly differ on the PEDI-CAT domains, except for daily activities (t(40) = -2.22, p = 0.037). Floor effects were significant for both measures, although the PEDI-CAT showed more floor effects in the mobility (35.7%) and social-cognitive (50%) domains. PEDI-CAT daily activities, mobility, social-cognitive and responsibility domains were all significantly correlated with most of the VABS-3 domains (all rho > 0.5; p < 0.01). Test-rest of the PEDI-CAT was comparable to the VABS-3. Results suggest that the PEDI-CAT is efficient, and minimal training is needed to administer it; however, it lacks specificity and shares a high rate of floor effects with the VABS-3.

Adaptive Skills in FXS: A Review of the Literature and Evaluation of the PEDI-Computer Adaptive Test (PEDI-CAT) to Measure Adaptive Skills.

Sleep quality, functional skills, and communication in preschool-aged children with autism spectrum disorder.

Abstract Introduction Children with chronic liver diseases are exposed to biological and/or environmental risk factors that can compromise their neuromotor acquisition and development of functional skills. Objective To describe the neuropsychomotor development (NPMD) and functional skills of children with chronic liver diseases. Methods Cross-sectional, descriptive and exploratory study carried out with children up to 6 years old who were selected at a reference hospital in the state of Bahia, Brazil, from November 2019 to March 2020. Children in outpatient care with clinical, laboratory and histological diagnosis compatible with chronic liver disease were considered eligible. The instrument for assessing neuropsychomotor development was Denver II. Functional skills were obtained by applying the Pediatric Evaluation of Disability Inventory computer-adaptive test (PEDI-CAT) to parents or primary caregivers, Speedy version (Speedy-CAT). Results Of the 34 children with chronic liver disease, 52.9% were female, aged between 4 and 6 years (64%). The results of the Denver II test showed that 68.7% (22/32) of the sample were at risk for NPMD. In the PEDI-CAT, the scores of children with liver disease at risk for NPMD were 60.7 ± 9.1 in the daily activity domains, 57.6 ± 11.8 in mobility and 48.3 ± 6.2 in the social/cognitive domains. Conclusion Children with chronic liver disease are at risk for NPMD, although not presenting impaired functional skills when evaluated by the PEDI-CAT.

Neuropsychomotor development and functional skills in preschool children with liver diseases

Resumo Introdução Crianças com doenças hepáticas crônicas são expostas a fatores de risco biológicos e/ou ambientais que podem comprometer suas aquisições neuromotoras e o desenvolvimento de suas habilidades funcionais. Objetivo Descrever o desenvolvimento neuropsicomotor (DNPM) e habilidades funcionais de crianças com doenças hepáticas crônicas. Métodos Estudo seccional, descritivo e exploratório realizado com crianças de até 6 anos, que foram selecionadas em um hospital de referência do estado da Bahia, Brasil, no período de novembro de 2019 a março de 2020. Foram consideradas elegíveis para o estudo crianças em atendimento ambulatorial, com diagnóstico clínico, laboratorial e histológico compatíveis com doença hepática crônica. O instrumento de avaliação do desenvolvimento neuropsicomotor foi o Denver II. As habilidades funcionais foram obtidas pela aplicação do Inventário de Avaliação Pediátrica de Incapacidade Testagem Computadorizada Adaptativa (PEDI-CAT) aos pais ou cuidadores principais, versão rápida (Speedy-CAT). Resultados Das 34 crianças com hepatopatias crônicas, 52,9% eram do sexo feminino, com idade entre 4 e 6 anos (64%). Os resultados do teste de Denver II demonstraram que 68,7% (22/32) da amostra apresentaram risco para DNPM. No PEDI-CAT, os escores das crianças hepatopatas com risco para DNPM foram de 60,7 ± 9,1 nos domínios atividade diária, 57,6 ± 11,8 em mobilidade e 48,3 ± 6,2 em social/cognitivo. Conclusão Crianças com hepatopatias crônicas apresentam risco para DNPM, apesar de não possuírem comprometimento de suas habilidades funcionais quando avaliadas pelo PEDI-CAT.

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Desenvolvimento neuropsicomotor e habilidades funcionais em pré-escolares com doenças hepáticas

AIM Assessment of functional abilities is an essential component in the diagnosis of neurodevelopmental disorders in young children. Functional abilities, assessed using the Pediatric Evaluation of Disability Inventory-Computer Adapted Test (PEDI-CAT), are used to determine eligibility for early intervention funded under the National Disability Insurance Scheme in Australia (NDIS). This study aims to compare the use of the PEDI-CAT with the Vineland-3, a comprehensive parent interview for the determination of support needs, diagnosis, and eligibility for funding. METHOD Seventy-five preschool aged children referred for formal diagnosis were assessed with the Vineland-3, and subscale scores reviewed for clinical judgement. These children were also assessed on the PEDI-CAT speedy version and the results on the two tests compared. RESULTS The PEDI-CAT was less sensitive than the Vineland-3 to the functional difficulties being experienced in this population of preschool aged children. The Vineland-3 had floor effects on some scales and deficits masked by skills in other areas in the same domain, but standard subscale scores allowed skills and areas needing support to be defined. Overall 25% of children clinically in need of support were not identified on the Pedi-cat compared to only 4% using Vineland Domain scores and 3% using Vineland-3 subscale scores. The PEDI-CAT also identified significantly lower levels of support required with only 32% of children found to need very substantial support compared to 40% of children on the Vineland Domain scores, and 59% using subscale scores. CONCLUSION Based on the results of this study, the Vineland-3 comprehensive interview interpreted using subscale scores as well as domain scores is more sensitive than the PEDI-CAT in identifying young children in need of support as well as the areas and degree of support required.

Lisa Campbell

Papers

Accurate assessment of functional abilities in pre-schoolers for diagnostic and funding purposes: A comparison of the Vineland-3 and the PEDI-CAT.