Prediction of clinical anxious and depressive problems in mid childhood amongst temperamentally inhibited preschool children: a population study.

Shy/inhibited young children are at risk for internalising difficulties; however, for many, this temperamental style does not result in mental health problems. This study followed a population-based sample of temperamentally inhibited preschool children into mid childhood to explore the aetiology of clinical-level anxious and depressive problems. Amongst inhibited preschool children, we aimed to predict each of clinical child anxiety and depressive problems in mid childhood from a broad range of potential risks (demographics, traumatic events and broader recent stressors, parents' well-being, and parenting practices). This study is based on data from a wider population trial of Cool Little Kids that recruited a representative sample of inhibited preschool children enrolled in their year before starting school. In 2011-2012, an inhibition screen was universally distributed to parents of children in their year before school (age 4 years) across eight diverse government areas in Melbourne, Australia. Participants were 545 parents of inhibited preschoolers (78% uptake, 545/703) who were followed to mid childhood (three annual waves 2015-2017, age 7-10 years) with 84% retention (456/545). Parents completed questionnaires spanning child ages 4-10 years, along with diagnostic interviews for child anxiety. Children also completed questionnaires in mid childhood. The questionnaires encompassed a variety of potential risks including sociodemographics, traumatic events, recent life stressors, parent wellbeing and parenting practices. In mid childhood, 57% (246/430) of inhibited preschoolers had a clinical level of anxiety problems while 22% (95/432) had depressive problems (by one or more sources). The aetiology analyses highlighted parent distress and parenting practices (overinvolved/protective, harsh discipline) as key predictors of inhibited preschoolers' internalising problems by mid childhood. Some high-risk families may not have participated. Child depression was not assessed with a diagnostic interview. The measures did not include every possible risk factor. The findings lend support to parenting programs for shy/inhibited young children that aim to prevent the development of anxiety and depression as they grow.

Follow-up of the Cool Little Kids translational trial into middle childhood.

BACKGROUND: Public health advocates have highlighted internalising problems as a leading cause of global burden of disease. Internalising problems (anxiety/depression) affect up to 20% of school-age children and can impact peer relations, school engagement and later employment and mortality. This translational trial aimed to determine whether a selective/indicated parenting group programme to prevent internalising distress in shy/inhibited preschool children had sustained effects in middle childhood. Translational design aspects were a brief parent-report screening tool for child inhibition offered universally across the population via preschools in the year before school, followed by an invitation to parents of all inhibited children to attend the parenting programme at venues in their local community. METHODS: Design of the study was a randomised controlled trial. The setting was 307 preschool services across eight socioeconomically diverse government areas in Melbourne, Australia. Participants were 545 parents of inhibited four-year-old children of which 456 (84%) were retained during middle childhood (age of seven to 10 years). Early intervention was the Cool Little Kids parenting group programme, and control was 'usual care' access to available support services in the community. Primary outcomes were child anxiety and depression symptoms (parent and child report) and DSM-IV anxiety disorders (assessor masked). Secondary outcomes were parenting practices and parent mental health. RESULTS: There was no significant difference in anxiety disorders between the intervention and control group during the three annual follow-ups of the cohort in middle childhood (2015 43% vs. 41%, 2016 40% vs. 36%, 2017 27% vs. 30%, respectively; p's > .05). There were also no significant differences in child anxiety or depression symptoms (by child or parent report), parenting practices or parent mental health, between the intervention and control group during middle childhood. However, a priori interaction tests suggested that for children with anxious parents, early intervention attenuated risk for middle childhood internalising problems. CONCLUSIONS: An issue for population translation is low levels of parent engagement in preventive interventions. Initial effects of the Cool Little Kids parenting group programme in reducing shy/inhibited preschool children's internalising distress at school entry dissipated over time, perhaps due to low engagement. Future translational research on early prevention of internalising problems could benefit from screening preschool children in the population at higher risk (combining temperamental inhibition and parent distress) and incorporating motivational techniques to facilitate family engagement. Trial registration ISRCTN30996662 http://www.isrctn.com/ISRCTN30996662.

Infant Regulation: Associations with Child Language Development in a Longitudinal Cohort.

OBJECTIVE: To determine whether infants who have regulatory problems (eg, sleeping, crying, and feeding problems) at 1 year of age are at increased risk of experiencing language difficulties at ages 5 and 11 years, compared with settled infants. STUDY DESIGN: Parent survey and child assessment data (n = 1131) were drawn from a longitudinal community cohort study. Latent Class Analysis identified 5 profiles of infant regulation including those who were settled (37%), had tantrums (21%), had sleep problems (25%), were moderately unsettled (13%), and severely unsettled (3%) at 12 months of age. Adjusted regression analyses examined associations between infant regulatory profiles and language ability (Clinical Evaluation of Language Fundamentals-fourth edition) at ages 5 and 11 years. RESULTS: Infants who were moderately unsettled had lower language scores at age 5 (adjusted mean difference, -3.89; 95% CI, -6.92 to -0.86) and were more likely to have language difficulties (aOR, 2.71; 95% CI, 1.28-5.75), than infants who were settled. Infants who were severely unsettled at 12 months of age, had lower language scores at ages 5 (adjusted mean difference, -7.71; 95% CI, -13.07 to -2.36) and 11 (adjusted mean difference, -6.50; 95% CI, -11.60 to -1.39), than infants who were settled. Severely unsettled infants were 5 times more likely to have language difficulties at age 5 than their settled counterparts (aOR, 5.01; 95% CI, 1.72-14.63). CONCLUSIONS: Children at 1 year of age with multiple regulatory problems are at an increased risk for poorer language skills at ages 5 and 11 years.

Cool Little Kids translational trial to prevent internalising: two-year outcomes and prediction of parent engagement.

BACKGROUND: The aim was to determine outcomes in the first year of school of a population-delivered parenting program to prevent internalising problems in temperamentally inhibited preschool children and predictors of engagement in parenting groups. METHOD: Design: Randomised controlled trial. SETTING: 307 preschool services across eight socio-economically diverse government areas in Melbourne, Australia. PARTICIPANTS: 545 parents of inhibited 4-year-old children; 469 (86%) retained at two-year follow-up. INTERVENTION: Cool Little Kids program. Primary outcomes were child internalising symptoms and anxiety disorders. Secondary outcomes were parenting, parent well-being and engagement. Trial registration ISRCTN30996662 http://www.isrctn.com/ISRCTN30996662. RESULTS: In the first year of school (M (SD) age 6.7 (0.4) years), child anxiety symptoms were reduced in the intervention versus control arm (PAS-R M (SD): total 36.2 (17.2) versus 39.4 (18.5); adjusted difference -3.26, 95% CI -6.46 to -0.05, p = .047; specific fears 9.1 (6.2) versus 10.7 (6.8), adjusted difference -1.53; 95% CI -2.69 to -0.38, p = .009). However, there was little difference in broader child internalising (CMFWQ M (SD): 2.2 (0.5) versus 2.3 (0.6); adjusted difference -0.03, 95% CI -0.13 to 0.06, p = .489) or anxiety disorders (37.6% vs. 42.6%; adjusted OR 0.79, 95% CI 0.53 to 1.18, p = .242). Lower income, younger mothers, less educated and more culturally diverse fathers engaged less with the intervention. Continued skills practice was less frequent for parents of girls and in advantaged neighbourhoods. CONCLUSIONS: There were population effects of Cool Little Kids in the first year of school for anxiety symptoms but not disorders. Considering motivation techniques to engage subgroups of families would be helpful in translation research.

DNA Methylation at Birth Predicts Intellectual Functioning and Autism Features in Children with Fragile X Syndrome.

Fragile X syndrome (FXS) is a leading single-gene cause of intellectual disability (ID) with autism features. This study analysed diagnostic and prognostic utility of the Fragile X-Related Epigenetic Element 2 DNA methylation (FREE2m) assessed by Methylation Specific-Quantitative Melt Analysis and the EpiTYPER system, in retrospectively retrieved newborn blood spots (NBS) and newly created dried blood spots (DBS) from 65 children with FXS (~2-17 years). A further 168 NBS from infants from the general population were used to establish control reference ranges, in both sexes. FREE2m analysis showed sensitivity and specificity approaching 100%. In FXS males, NBS FREE2m strongly correlated with intellectual functioning and autism features, however associations were not as strong for FXS females. Fragile X mental retardation 1 gene (FMR1) mRNA levels in blood were correlated with FREE2m in both NBS and DBS, for both sexes. In females, DNAm was significantly increased at birth with a decrease in childhood. The findings support the use of FREE2m analysis in newborns for screening, diagnostic and prognostic testing in FXS.

Associations between early maternal behaviours and child language at 36 months in a cohort experiencing adversity.

BACKGROUND: Variations in parenting, more specifically less responsive and more directive parenting, contribute to language difficulties for children experiencing adversity. Further investigation of associations between specific responsive and directive behaviours and child language is required to understand how behaviours shape language over time within different populations. As language is dyadic, further exploration of how mother-child interactions moderate associations is also important. AIMS: To investigate associations between specific responsive and directive maternal behaviours, the quality of mother-child interaction (fluency and connectedness) and child language in a cohort experiencing adversity. METHODS & PROCEDURES: Pregnant women experiencing adversity were recruited from maternity hospitals in Australia. At 12 months, videos of mother-infant free play were collected. Videos were coded for maternal behaviours and fluency and connectedness (n = 249). At 36 months, child language was measured using a standardized language test. Linear regression models were used to examine associations and the moderating role of fluency and connectedness was explored. OUTCOMES & RESULTS: Responsive yes/no questions were positively associated with language scores. Unsuccessful redirectives were negatively associated with language scores. The moderation effect of fluency and connectedness was equivocal in the current data. CONCLUSIONS & IMPLICATIONS: Findings reproduce and extend previous research highlighting key features of mother-child interactions associated with child language trajectories. Findings also augment knowledge of risk and protective factors related to language for children experiencing adversity and highlight where targeted interventions might be successful.

Significantly Elevated FMR1 mRNA and Mosaicism for Methylated Premutation and Full Mutation Alleles in Two Brothers with Autism Features Referred for Fragile X Testing.

Although fragile X syndrome (FXS) is caused by a hypermethylated full mutation (FM) expansion with ≥200 cytosine-guanine-guanine (CGG) repeats, and a decrease in FMR1 mRNA and its protein (FMRP), incomplete silencing has been associated with more severe autism features in FXS males. This study reports on brothers (B1 and B2), aged 5 and 2 years, with autistic features and language delay, but a higher non-verbal IQ in comparison to typical FXS. CGG sizing using AmplideX PCR only identified premutation (PM: 55-199 CGGs) alleles in blood. Similarly, follow-up in B1 only revealed PM alleles in saliva and skin fibroblasts; whereas, an FM expansion was detected in both saliva and buccal DNA of B2. While Southern blot analysis of blood detected an unmethylated FM, methylation analysis with a more sensitive methodology showed that B1 had partially methylated PM alleles in blood and fibroblasts, which were completely unmethylated in buccal and saliva cells. In contrast, B2 was partially methylated in all tested tissues. Moreover, both brothers had FMR1 mRNA ~5 fold higher values than those of controls, FXS and PM cohorts. In conclusion, the presence of unmethylated FM and/or PM in both brothers may lead to an overexpression of toxic expanded mRNA in some cells, which may contribute to neurodevelopmental problems, including elevated autism features.

Translational delivery of Cool Little Kids to prevent child internalising problems: Randomised controlled trial.

OBJECTIVE: To determine whether a population-delivered parenting programme assists in preventing internalising problems at school entry for preschool children at-risk with temperamental inhibition. METHODS: Design: a randomised controlled trial was used. SETTING: the setting was 307 preschool services across eight socioeconomically diverse government areas in Melbourne, Australia. PARTICIPANTS: a total of 545 parents of inhibited 4-year-old children: 498 retained at 1-year follow up. Early intervention: Cool Little Kids parenting group programme was implemented. Primary outcomes: the primary outcomes were child DSM-IV anxiety disorders (assessor blind) and internalising problems. SECONDARY OUTCOMES: the secondary outcomes were parenting practices and parent mental health. RESULTS: At 1-year follow up (mean (standard deviation) age = 5.8 (0.4) years), there was little difference in anxiety disorders between the intervention and control arms (44.2% vs 50.2%; adjusted odds ratio = 0.86, 95% confidence interval = [0.60, 1.25], p = 0.427). Internalising problems were reduced in the intervention arm (Strengths and Difficulties Questionnaire: abnormal - 24.2% vs 33.0%; adjusted odds ratio = 0.56, 95% confidence interval = [0.35, 0.89], p = 0.014; symptoms - mean (standard deviation) = 2.5 (2.0) vs 2.9 (2.2); adjusted mean difference = -0.47, 95% confidence interval = [-0.81, -0.13], p = 0.006). Parents' participation in the intervention was modest (29.4% attended most groups, 20.5% used skills most of the time during the year). A priori interaction tests suggested that for children with anxious parents, the intervention reduced anxiety disorders and internalising symptoms after 1 year. CONCLUSION: Offering Cool Little Kids across the population for inhibited preschoolers does not impact population outcomes after 1 year. Effects may be emerging for inhibited children at highest risk with parent anxiety. Trial outcomes will continue into mid-childhood.

Language and social-emotional and behavioural wellbeing from 4 to 7 years: a community-based study.

Language disorder (LD) and social-emotional and behavioural (SEB) difficulties are common childhood problems that often co-occur. While there is clear evidence of these associations from clinical samples, less is known about community samples. This paper examines these associations in children aged 4-7 years from a community-based longitudinal study. 771 families provided questionnaire and assessment data at 4, 5 and 7 years. Parent-reported SEB difficulties were measured at each point (SDQ). Child language was directly assessed at 4 (CELF-P2), 5 and 7 years (CELF-4). Linear regression analysis was used to compare cross-sectional differences in mean SDQ scores between children with and without LD at each time point. Linear regression was then used to examine how patterns of language development (language disordered at three time points; never disordered; disordered at one or two time points, i.e. 'unstable' group) related to SEB difficulties at each age, adjusted for potential confounders, as in the previous analyses. Higher hyperactivity/inattention scores were associated with LD at each age. In fully adjusted models, there was little difference in mean emotional symptoms scores between children with and without LD. The 'never' LD group had lower mean SDQ scores at each time point than the 'unstable' group. Findings highlight that children with persistent LD from preschool to early primary school may be more likely to have concomitant SEB difficulties, particularly behavioural difficulties. Those with unstable LD may also have co-occurring SEB difficulties, showing a need for education and health professionals to monitor early language and SEB development.

Predictive validity of verbal and non-verbal communication and mother-child turn-taking at 12 months on language outcomes at 24 and 36 months in a cohort of infants experiencing adversity: a preliminary study.

BACKGROUND: Parent-reported measures of early communication have limitations for use with infants experiencing adversity. Observational measures of early non-verbal and verbal communicative behaviours and mother-child turn-taking may provide a complementary method of capturing early communication skills for these children. AIMS: To explore the predictive validity of verbal and non-verbal behaviours and mother-child conversational turn-taking (fluency and connectedness) at child age 12 months in relation to language measures at 24 and 36 months in a cohort of infants experiencing adversity. METHODS & PROCEDURES: Pregnant women experiencing adversity were recruited from maternity hospitals in Australia. At 12 months, 190 infants were videoed during mother-child free-play. Verbal and non-verbal communicative behaviours and fluency and connectedness were measured from the 12-month videos. Predictive validity of 12-month behaviours was calculated in relation to mean length of utterance and number of unique words at 24 months and Clinical Evaluation of Language Fundamentals Preschool-Second Edition (CELF-P2) Core Language scores at 36 months. OUTCOMES & RESULTS: All 12-month behaviours had adequate specificity but poor sensitivity when compared with other predictive validity studies using published early language measures. However, in adjusted regression models, fluency and connectedness and verbal behaviours at 12 months predicted unique words at 24 months. Fluency and connectedness also predicted CELF-P2 scores at 36 months. CONCLUSIONS & IMPLICATIONS: Findings reconfirm the difficulty in early identification of children at risk of later language difficulties. All 12-month measures were more accurate at identifying those children who will have better language than those children who will not. As fluency and connectedness was the only measure to predict 24- and 36-month language in adjusted regression models, it may be an important factor to consider when measuring early language skills for infants experiencing adversity. Future research could combine observational measures of early communication and fluency and connectedness with other predictors of language to try to increase prediction accuracy.

Partially methylated alleles, microdeletion, and tissue mosaicism in a fragile X male with tremor and ataxia at 30 years of age: A case report.

CGG repeat expansion >200 within FMR1, termed full mutation (FM), has been associated with promoter methylation, consequent silencing of gene expression and fragile X syndrome (FXS)-a common cause of intellectual disability and co-morbid autism. Unmethylated premutation (55-199 repeats) and FM alleles have been associated with fragile X related tremor/ataxia syndrome (FXTAS), a late onset neurodegenerative disorder. Here we present a 33-year-old male with FXS, with white matter changes and progressive deterioration in gait with cerebellar signs consistent with probable FXTAS; there was no evidence of any other cerebellar pathology. We show that he has tissue mosaicism in blood, saliva, and buccal samples for the size and methylation of his expanded alleles and a de novo, unmethylated microdeletion. This microdeletion involves a ∼80 bp sequence in the FMR1 promoter as well as complete loss of the CGG repeat in a proportion of cells. Despite FMR1 mRNA levels in blood within the normal range, the methylation and CGG sizing results are consistent with the diagnosis of concurrent FXS and probable FXTAS. The demonstrated presence of unmethylated FM alleles would explain the manifestation of milder than expected cognitive and behavioral impairments and early onset of cerebellar ataxia. Our case suggests that individuals with FXS, who manifest symptoms of FXTAS, may benefit from more detailed laboratory testing. © 2016 Wiley Periodicals, Inc.

Early detection of fragile X syndrome: applications of a novel approach for improved quantitative methylation analysis in venous blood and newborn blood spots.

BACKGROUND: Standard fragile X syndrome (FXS) diagnostic tests that target methylation of the fragile X mental retardation 1 (FMR1) CpG island 5' of the CGG expansion can be used to predict severity of the disease in males from birth, but not in females. METHODS: We describe methylation specific-quantitative melt analysis (MS-QMA) that targets 10 CpG sites, with 9 within FMR1 intron 1, to screen for FXS from birth in both sexes. The novel method combines the qualitative strengths of high-resolution melt and the high-throughput, quantitative real-time PCR standard curve to provide accurate quantification of DNA methylation in a single assay. Its performance was assessed in 312 control (CGG <40), 143 premutation (PM) (CGG 56-170), 197 full mutation (FM) (CGG 200-2000), and 33 CGG size and methylation mosaic samples. RESULTS: In male and female newborn blood spots, MS-QMA differentiated FM from control alleles, with sensitivity, specificity, and positive and negative predictive values between 92% and 100%. In venous blood of FM females between 6 and 35 years of age, MS-QMA correlated most strongly with verbal IQ impairment (P = 0.002). In the larger cohort of males and females, MS-QMA correlated with reference methods Southern blot and MALDI-TOF mass spectrometry (P < 0.05), but was not significantly correlated with age. Unmethylated alleles in high-functioning FM and PM males determined by both reference methods were also unmethylated by MS-QMA. CONCLUSIONS: MS-QMA has an immediate application in FXS diagnostics, with a potential use of its quantitative methylation output for prognosis in both sexes.

Pragmatic language difficulties in children with hyperactivity and attention problems: an integrated review.

BACKGROUND: Diagnostic criteria for attention deficit hyperactivity disorder (ADHD) suggest a range of difficulties in the pragmatic aspects of language, including excessive talking and interrupting others. Such difficulties have been periodically reported over several decades in studies on the language abilities of children with features of ADHD, yet a comprehensive review of the literature has been lacking. AIMS: This review aims to integrate evidence from several lines of research from 1979 to the present on pragmatic language difficulties in children with ADHD or symptoms of ADHD. METHODS & PROCEDURES: A comprehensive search of empirical literature on pragmatic language in children with ADHD or symptoms of ADHD was conducted using PsycINFO and PubMed databases and through following up relevant references cited in articles. Literature was reviewed with respect to the nature and extent of pragmatic language difficulties in ADHD. OUTCOMES & RESULTS: Thirty studies met the review inclusion criteria, including recent questionnaire studies, observational studies of children's communication patterns, and studies of higher-level language comprehension and production. The studies indicate a consistent profile of pragmatic language impairments in children with features of ADHD, particularly in the areas of excessive talking, poor conversational turn-taking, and lack of coherence and organization in elicited speech. CONCLUSIONS & IMPLICATIONS: Pragmatic language difficulties are common in children with features of ADHD. These difficulties are consistent with deficits in executive function that are thought to characterize ADHD, thus providing some support for the theory that executive function contributes to pragmatic language competency. As yet there is very little empirical evidence of specific relationships between particular aspects of pragmatic language and particular domains of executive function. Given the importance of pragmatic language competency for children's social and academic functioning, pragmatic language abilities should be considered during clinical assessment for ADHD and targeted for intervention.

Stability of language performance at 4 and 5 years: measurement and participant variability.

BACKGROUND: Language impairment (LI) in the preschool years is known to vary over time. Stability in the diagnosis of LI may be influenced by children's individual variability, the measurement error of commonly used assessment instruments and the cut-points used to define impairment. AIMS: To investigate the agreement between two different age-based versions of a language assessment instrument and the stability of the classification of LI using the two measures over a 12-month period. METHODS & PROCEDURES: A total of 945 participants completed the Clinical Evaluation of Language Fundamentals(CELF­Preschool 2 or 4th Edn) at 4 and 5 years of age. Agreement and stability were analysed using Bland­Altman plots, correlation and odds ratios. Sensitivity and specificity were calculated for two thresholds of the CELF-P2 using the diagnostic category on the child's subsequent CELF-4. OUTCOMES & RESULTS: For all CELF scores, mean differences for the cohort between 4 and 5 years were within 1.5 scale score units. In contrast, at the individual level variability was found across the range of scores and was of a greater magnitude than previously reported. Stability in LI classification was low, with 36% of 5-year-olds with LI (defined as a standard score below ­1.25) classified as typical at 4 years, even though odds ratios calculated from classifications at the two time points suggested that 4-year-olds with LI had 23 times greater odds than their typical peers to receive a diagnosis of LI at 5 years. The CELF-P2 did not demonstrate adequate levels of diagnostic accuracy for LI at 5 years: sensitivity of 64% and specificity of 92.9%. CONCLUSIONS: Substantial variability across the entire range of possible CELF scores was observed in this community cohort between the ages of 4 and 5 years. The stability of LI classification was lower than that reported in previous research conducted primarily on smaller clinical cohorts. The current study's results suggest that the variability observed in developmental language pathways is the result of a combination of limitations in measurement instruments, individual children's abilities and the arbitrary nature of the boundaries defining LI.

Computer use and letter knowledge in pre-school children: a population-based study.

AIM: To explore the link between pre-school children's general home computer use and their letter knowledge. METHODS: As part of the Early Language in Victoria Study, a community cohort of 1539 four-year-old children was tested on letter knowledge as well as on non-verbal intelligence, oral language, articulation and phonological awareness. Performance on these measures was examined in relation to parent-questionnaire responses exploring home literacy environment and the amount of time children spent using the computer. RESULTS: A positive correlation between computer use and letter knowledge was found, and this association was still evident after controlling for other cognitive and environmental factors known to predict the development of letter knowledge in young children. CONCLUSIONS: Greater computer use in pre-school children appears to have a positive association with emerging literacy development. Future research needs to examine the nature of that association.

Validity of the Children's Moods, Fears and Worries Questionnaire in a clinical setting.

BACKGROUND: The Children's Moods, Fears and Worries Questionnaire (CMFWQ) is a recently developed parent-report measure for detecting young children's internalising difficulties. This study evaluated construct and criterion-related validity with a clinical sample. METHOD: The measure was completed by 30 mothers of children aged between 18 months and 7 years referred to hospital clinical psychology services. RESULTS: The CMFWQ demonstrated sound convergent and discriminant validity and a clinical cut-point was established, which optimally discriminated between children with internalising problems and those within the normal range. CONCLUSION: The CMFWQ has potential value for identifying young children in need of intervention for internalising problems.

Agreement between parents' and clinical researchers' ratings of behavioral problems in children with fragile X syndrome and chromosome 15 imprinting disorders.

BACKGROUND: Despite the increasing number of clinical trials involving children with neurodevelopmental disorders, appropriate and objective outcome measures for behavioral symptoms are still required. AIM: This study assessed the agreement between parents' and clinical researchers' ratings of behavioral problem severity in children with fragile X syndrome (FXS) and chromosome 15 imprinting disorders. METHODS AND PROCEDURES: The cohort comprised 123 children (64% males), aged 3-17 years, with FXS (n = 79), Prader-Willi (PWS; n = 19), Angelman (AS; n = 15), and Chromosome 15q duplication (n = 10) syndromes. Specific items from the Autism Diagnostic Observation Schedule-Second Edition and Aberrant Behavior Checklist-Community Edition mapping to corresponding behavioral domains were selected ad-hoc, to assess behavioral problems. OUTCOMES AND RESULTS: Inter-rater agreement for the cohort was slight for self-injury (Intraclass Correlation Coefficient (ICC) = 0.12), fair for tantrums/aggression (0.24) and mannerisms/stereotypies (0.25), and moderate for hyperactivity (0.48). When stratified by diagnosis, ICC ranged from poor (0; self-injury, AS and PWS) to substantial (0.48; hyperactivity, females with FXS). CONCLUSIONS AND IMPLICATIONS: The high level of inter-rater disagreement across most domains suggests that parents' and researchers' assessments led to discrepant appraisal of behavioral problem severity. These findings have implications for treatment targets and outcome measure selection in clinical trials, supporting a multi-informant approach.

Predictors in Infancy for Language and Academic Outcomes at 11 Years.

OBJECTIVES: To examine the contribution of early life factors and preschool- and school-aged language abilities to children's 11-year language and academic outcomes. METHODS: Participants (N = 839) were from a prospective community cohort study of 1910 infants recruited at 8 to 10 months of age. Early life factors included a combination of child (prematurity, birth weight), family (socioeconomic disadvantage, family history of language difficulties), and maternal factors (education, vocabulary, and age). Language (standardized assessment of receptive and expressive skills) and academic (national assessment) outcomes at 11 years were predicted by using a series of multivariable regression models. RESULTS: Early life factors explained 11% to 12% of variance in language scores at 11 years. The variance explained increased to 47% to 64% when language scores from 2 to 7 years were included. The largest increase in variance explained was with 4-year language scores. The same early life factors explained 13% to 14% of academic scores at 11 years, with increases to 43% to 54% when language scores from 2 to 11 years were included. Early life factors adequately discriminated between children with typical and low language scores but were much better discriminators of children with typical and low academic scores. When earlier language scores were added to models then the area under the curve increased to 0.9 and above. CONCLUSIONS: Children's language outcomes at 11 years are accurately predicted by their 4-year language ability and their academic outcomes at 11 years are predicted by early family and home environment factors. Children with low language abilities at 11 years consistently performed more poorly on national assessments of literacy and numeracy.

Assessing early communication behaviours: structure and validity of the Communication and Symbolic Behaviour Scales-Developmental Profile (CSBS-DP) in 12-month-old infants.

BACKGROUND: Parent report instruments are frequently used for the identification of both 'at-risk' children and to support the diagnosis of communication delay. Whilst the evidence is strong for the accuracy of parent report of vocabulary between 2 and 3 years, there are fewer studies that have considered the ability of parents to report on early communication behaviours in 12-month-old infants. AIMS: To investigate the validity of the underlying structure of the Communication and Symbolic Behaviour Scales-Developmental Profile (CSBS-DP) for each of the direct observation and parent reports of communication behaviour in infants at 12 months of age. METHODS & PROCEDURES: Participants were 1725 infants, already participating in a longitudinal study of language development, whose parents completed the Infant-Toddler Checklist from the CSBS-DP. Seven hundred and twenty-eight (728) of these infants also completed the Behaviour Sample from the CSBS-DP. The structure of the CSBS-DP was examined using confirmatory factor analyses (CFA) of the Behaviour Sample and the Infant-Toddler Checklist. Correlations between the Infant-Toddler Checklist and the Behaviour Sample on the total, composite, and subscale scores were also calculated. OUTCOMES & RESULTS: Confirmatory factor analysis of the CSBS-DP Behaviour Sample replicated previous work conducted during the development of the instrument, but on a larger and younger cohort of Australian infants. The data provided support for at least three factors, broadly representing Social, Speech, and Symbolic communication skills, with some evidence that the speech factor could be further split into sub-factors representing Sounds and Words. There was support for a three-factor structure for the Infant-Toddler Checklist. Moderate correlations were found between results from the Behaviour Sample and the Infant-Toddler Checklist. CONCLUSIONS & IMPLICATIONS: As measures of early communication skill for young infants, the CSBS-DP Behaviour Sample and the Infant-Toddler Checklist are valid clinical tools for measuring constructs broadly representing Social, Speech, and Symbolic communication skills. The Speech composite in particular emerged as a strong factor under the Behaviour Sample.

Predictors of early precocious talking: a prospective population study.

This study examines potential predictors of 'precocious talking' (expressive language ≥90th percentile) at one and two years of age, and of 'stability' in precocious talking across both time periods, drawing on data from a prospective community cohort comprising over 1,800 children. Logistic regression was used to examine the relationship between precocious talking and the following potential predictors: gender, birth order, birth weight, non-English speaking background, socioeconomic status, maternal age, maternal mental health scores, and vocabulary and educational attainment of parents. The strongest predictors of precocity (being female and having a younger mother) warrant further exploration. Overall, however, it appears that precocity in early vocabulary development is not strongly influenced by the variables examined, which together explained just 2.6% and 1% of the variation at 1;0 and 2;0 respectively.