Early moderate prenatal alcohol exposure and maternal diet impact offspring DNA methylation across species.

Alcohol consumption in pregnancy can affect genome regulation in the developing offspring but results have been contradictory. We employed a physiologically relevant murine model of short-term moderate prenatal alcohol exposure (PAE) resembling common patterns of alcohol consumption in pregnancy in humans. Early moderate PAE was sufficient to affect site-specific DNA methylation in newborn pups without altering behavioural outcomes in adult littermates. Whole-genome bisulfite sequencing of neonatal brain and liver revealed stochastic influence on DNA methylation that was mostly tissue-specific, with some perturbations likely originating as early as gastrulation. DNA methylation differences were enriched in non-coding genomic regions with regulatory potential indicative of broad effects of alcohol on genome regulation. Replication studies in human cohorts with fetal alcohol spectrum disorder suggested some effects were metastable at genes linked to disease-relevant traits including facial morphology, intelligence, educational attainment, autism, and schizophrenia. In our murine model, a maternal diet high in folate and choline protected against some of the damaging effects of early moderate PAE on DNA methylation. Our studies demonstrate that early moderate exposure is sufficient to affect fetal genome regulation even in the absence of overt phenotypic changes and highlight a role for preventative maternal dietary interventions.

Drinking excessive amounts of alcohol during pregnancy can cause foetal alcohol spectrum disorder and other conditions in children that affect their physical and mental development. Many countries advise women who are pregnant or trying to conceive to avoid drinking alcohol entirely. However, surveys of large groups of women in Western countries indicate that most women continue drinking low to moderate amounts of alcohol until they discover they are pregnant and then stop consuming alcohol for the rest of their pregnancy. It remains unclear how this common drinking pattern affects the foetus. The instructions needed to build and maintain a human body are stored within molecules of DNA. Some regions of DNA called genes contain the instructions to make proteins, which perform many tasks in the body. Other so-called 'non-coding' regions do not code for any proteins but instead have roles in regulating gene activity. One way cells control which genes are switched on or off is adding or removing tags known as methyl groups to certain locations on DNA. Previous studies indicate that alcohol may affect how children develop by changing the patterns of methyl tags on DNA. To investigate the effect of moderate drinking during the early stages of pregnancy, Bestry et al. exposed pregnant mice to alcohol and examined how this affected the patterns of methyl tags on DNA in their offspring. The experiments found moderate levels of alcohol were sufficient to alter the patterns of methyl tags in the brains and livers of the newborn mice. Most of the changes were observed in non-coding regions of DNA, suggesting alcohol may affect how large groups of genes are regulated. Fewer changes in the patterns of methyl tags were found in mice whose mothers had diets rich in two essential nutrients known as folate and choline. Further experiments found that some of the affected mouse genes were similar to genes linked to foetal alcohol spectrum disorder and other related conditions in humans. These findings highlight the potential risks of consuming even moderate levels of alcohol during pregnancy and suggest that a maternal diet rich in folate and choline may help mitigate some of the harmful effects on the developing foetus.

An evaluation of a multi-site fetal alcohol spectrum disorder models of care project.

Fetal alcohol spectrum disorder (FASD) continues to be underdiagnosed in Australia, partly due to the lack of trained clinicians and diagnostic services. This project aimed to help increase FASD knowledge and diagnostic capacity across Australia. Six sites across Australia formed part of a national consortium, delivering training clinics, diagnostic clinics and community education sessions. The number of FASD diagnoses significantly increased across the project. Additionally, the number of community education sessions steadily increased across the project, with largely positive feedback. Participants attending the training clinics demonstrated increased knowledge of and confidence in FASD diagnosis. This evaluation showcases the benefits of a coordinated approach to prevention, assessment, diagnosis and training in FASD.

Scoping review of employment resources for individuals with fetal alcohol spectrum disorder and justice involvement.

OBJECTIVES: Individuals with fetal alcohol spectrum disorder (FASD) are over-represented within the justice system and have significant employment challenges. The primary aim of this scoping review was to ascertain available employment resources for FASD individuals particularly those involved in the justice system. Secondary aims were to determine available evidence-based interventions for the justice workforce and employment providers. METHODS: Eligibility criteria: That the resource was (a) published between 1990 and 2021, (b) in English, (c) available electronically in full text, (d) focused on strategies for improving employment outcomes of individuals with FASD and (f) developed for those aged over 15. Sources of evidence: Electronic searches of the following databases were conducted: EMBASE, MEDLINE, PsycINFO, Scopus, Web of Science and Google Scholar. Grey literature was collected via the databases ProQuest Dissertations & Theses Global, OpenGrey, GreyNet International and Grey Matters. Charting methods: Using Preferred Reporting Items for Systematic Reviews and Meta-Analyses extension for Scoping Reviews reporting guidelines, a five-stage methodological framework was employed. A quality appraisal of identified resources was conducted. Data were summarised qualitatively using a content analysis method that allowed for analysis of specific terms/themes/concepts/resource elements that resources covered, as well as a quantitative analysis of their frequency. RESULTS: An initial search identified 850 articles, 512 of which were obtained through online database searches, 321 through Google Scholar searches and 17 through google searches. Sixteen resources were identified as meeting inclusion criteria, including four peer-reviewed papers and 12 grey literature resources. Six of the resources extracted were deemed 'good' or 'strong' quality, with the remainder-all grey literature resources-being 'adequate' or 'limited'. None of the resources identified were empirically evaluated or could be implemented synergistically. Content analysis revealed common themes addressing FASD-related employment challenges and required supports. CONCLUSIONS: This review highlights the need for developing evidence-based employment-related resources for justice-involved people with FASD. Most of the 16 identified resources had a psychoeducational and advocacy component and described typical employment challenges with specific supports and accommodations to maximise workforce participation. However, none had been empirically evaluated, underscoring the importance of future research in this area to better inform more responsive and evidence-based employment-related programmes. TRIAL REGISTRATION NUMBER: doi:10.17605/OSF.IO/J5VMB.

A reduction in reported alcohol use in pregnancy in Australian Aboriginal communities: a prevention campaign showing promise.

OBJECTIVE: Aboriginal leaders in remote Western Australian communities with high rates of prenatal alcohol exposure invited researchers to evaluate the community-led Marulu foetal alcohol spectrum disorder (FASD) Prevention Strategy initiated in 2010. METHODS: The proportion of women reporting alcohol use during pregnancy to midwives was compared between 2008, 2010 and 2015. Initial midwife appointments were calculated by weeks of gestation. The proportions of women reporting alcohol use by age at birth were compared. RESULTS: Alcohol use reduced significantly from 2010 (61.0%) to 2015 (31.9%) with first-trimester use reducing significantly from 2008 (45.1%) to 2015 (21.6%). Across all years, 40.8% reported alcohol use during pregnancy and 14.8% reported use in both first and third trimesters. Most women attended the midwife in the first trimester. There was a significant relationship between increased maternal age and third-trimester alcohol use. CONCLUSIONS: The reduction in reported prenatal alcohol exposure in an Aboriginal community setting during a period of prevention activities provides initial evidence for a community-led strategy that might be applicable to similar communities. Implications for public health: The reductions in alcohol use reduce the risk of children being born with FASD in an area with high prevalence, with possible resultant reductions in associated health, economic and societal costs.

School-based intervention to address self-regulation and executive functioning in children attending primary schools in remote Australian Aboriginal communities.

Executive functioning and self-regulation influence a range of outcomes across the life course including physical and mental health, educational success, and employment. Children prenatally exposed to alcohol or early life trauma (ELT) are at higher risk of impairment of these skills and may require intervention to address self-regulation deficits. Researchers partnered with the local Aboriginal health organization and schools to develop and pilot a manualized version of the Alert Program® in the Fitzroy Valley, north Western Australia, a region with documented high rates of fetal alcohol spectrum disorder and ELT. This self-controlled cluster randomized trial evaluated the effect of an 8-week Alert Program® intervention on children's executive functioning and self-regulation skills. Following parent or caregiver consent (referred to hereafter as parent), 271 students were enrolled in the study. This reflects a 75% participation rate and indicates the strong community support that exists for the study. Teachers from 26 primary school classrooms across eight Fitzroy Valley schools received training to deliver eight, one-hour Alert Program® lessons over eight-weeks to students. Student outcomes were measured by parent and teacher ratings of children's behavioral, emotional, and cognitive regulation. The mean number of lessons attended by children was 4.2. Although no significant improvements to children's executive functioning skills or behavior were detected via the teacher-rated measures as hypothesized, statistically significant improvements were noted on parent-rated measures of executive functioning and behavior. The effectiveness of future self-regulation programs may be enhanced through multimodal delivery through home, school and community based settings to maximize children's exposure to the intervention. Despite mixed findings of effect, this study was an important first step in adapting and evaluating the Alert Program® for use in remote Australian Aboriginal community schools, where access to self-regulation interventions is limited.

The Demographic and Neurocognitive Profile of Clients Diagnosed With Fetal Alcohol Spectrum Disorder in PATCHES Paediatrics Clinics Across Western Australia and the Northern Territory.

BACKGROUND: Fetal alcohol spectrum disorder (FASD) is a diagnosis relating to neurocognitive impairments associated with prenatal alcohol exposure. A key aspect of improving FASD diagnostic processes and management is understanding the demographic and neurocognitive profile of those living with FASD. The aim of this study was to describe the demographic and neurocognitive profile of the first 199 individuals diagnosed with FASD in PATCHES Paediatrics clinics. METHODS: A retrospective cross-sectional descriptive study design was conducted with individuals diagnosed with FASD between 2013 and 2018 through a multidisciplinary team according to the Australian FASD Diagnostic Guidelines. RESULTS: Participants were primarily male 133 (66.8%) and Aboriginal Australian 147 (73.9%), aged 2 to 31 (mean 10.5), with 94 (47.3%) from remote or very remote parts of Western Australia. Participants came from low 119 (59.8%), medium 48 (24.1%), and high 32 (16.1%) socioeconomic (SE) backgrounds. Low SE background was found to be a predictor of number of sentinel facial features (Wald χ2 (1) = 4.03, p < 0.05). Most received a diagnosis of FASD with <3 sentinel features 165 (82.9%). Participants either had 6 or more 46 (23.1%), 5 44 (22.1%), 4 55 (27.6%), or 3 (27.1%) neurodevelopmental domains impaired. Executive functioning was the most commonly impaired neurodevelopmental domain 158 (79.4%), and 31 (61%) reported sleep disturbance. ADHD was the most observed comorbid condition (41.7%). CONCLUSIONS: This study improves our current understanding of neurocognitive and demographic profiles in individuals with FASD that have been clinically referred for diagnosis within Western Australia and the Northern Territory, and highlights the importance of prevention and early assessment/diagnosis as well as guidance regarding more targeted interventions. FASD affects individuals from all cultural and SE backgrounds. Individuals from middle to higher SE groups are at risk of FASD with prevention efforts needing to target these sectors of society. Suggestions for future research directions are also provided.

Study protocol for a self-controlled cluster randomised trial of the Alert Program to improve self-regulation and executive function in Australian Aboriginal children with fetal alcohol spectrum disorder.

INTRODUCTION: While research highlights the benefits of early diagnosis and intervention for children with fetal alcohol spectrum disorders (FASD), there are limited data documenting effective interventions for Australian children living in remote communities. METHODS AND ANALYSIS: This self-controlled cluster randomised trial is evaluating the effectiveness of an 8-week Alert Program school curriculum for improving self-regulation and executive function in children living in remote Australian Aboriginal communities. Children in grades 1-6 attending any of the eight participating schools across the Fitzroy Valley in remote North-West Australia (N ≈ 363) were invited to participate. Each school was assigned to one of four clusters with clusters randomly assigned to receive the intervention at one of four time points. Clusters two, three and four had extended control conditions where students received regular schooling before later receiving the intervention. Trained classroom teachers delivered the Alert Program to students in discrete, weekly, 1-hour lessons. Student outcomes were assessed at three time points. For the intervention condition, data collection occurred 2 weeks immediately before and after the intervention, with a follow-up 8 weeks later. For control conditions in clusters two to four, the control data collection matched that of the data collection for the intervention condition in the preceding cluster. The primary outcome is change in self-regulation. FASD diagnoses will be determined via medical record review after the completion of data collection. The results will be analysed using generalised linear mixed modelling and reported in accordance with Consolidated Standards of Reporting Trials (CONSORT) guidelines. ETHICS AND DISSEMINATION: Ethical approval was obtained from the University of Western Australia (WA) (RA/4/1/7234), WA Aboriginal Health Ethics Committee (601) and WA Country Health Service (2015:04). The Kimberley Aboriginal Health Planning Forum Research Sub-Committee and WA Department of Education also provided approval. The results will be disseminated through peer-reviewed journals, conference presentations, the media and at forums. TRIAL REGISTRATION NUMBER: ACTRN12615000733572; Pre-results.

Fine motor skills in a population of children in remote Australia with high levels of prenatal alcohol exposure and Fetal Alcohol Spectrum Disorder.

BACKGROUND: Many children in the remote Fitzroy Valley region of Western Australia have prenatal alcohol exposure (PAE). Individuals with PAE can have neurodevelopmental impairments and be diagnosed with one of several types of Fetal Alcohol Spectrum Disorder (FASD). Fine motor skills can be impaired by PAE, but no studies have developed a comprehensive profile of fine motor skills in a population-based cohort of children with FASD. We aimed to develop a comprehensive profile of fine motor skills in a cohort of Western Australian children; determine whether these differed in children with PAE or FASD; and establish the prevalence of impairment. METHODS: Children (n = 108, 7 to 9 years) were participants in a population-prevalence study of FASD in Western Australia. Fine motor skills were assessed using the Bruininks-Oseretsky Test of Motor Proficiency, which provided a Fine Motor Composite score, and evaluated Fine Manual Control (Fine Motor Precision; Fine Motor Integration) and Manual Coordination (Manual Dexterity; Upper-Limb Coordination). Descriptive statistics were reported for the overall cohort; and comparisons made between children with and without PAE and/or FASD. The prevalence of severe (≤ 2nd percentile) and moderate (≤16th percentile) impairments was determined. RESULTS: Overall, Fine Motor Composite scores were 'average' (M = 48.6 ± 7.4), as were Manual Coordination (M = 55.7 ± 7.9) and Fine Manual Control scores (M = 42.5 ± 6.2). Children with FASD had significantly lower Fine Motor Composite (M = 45.2 ± 7.7 p = 0.046) and Manual Coordination scores (M = 51.8 ± 7.3, p = 0.027) than children without PAE (Fine Motor Composite M = 49.8 ± 7.2; Manual Coordination M = 57.0 ± 7.7). Few children had severe impairment, but rates of moderate impairment were very high. CONCLUSIONS: Different types of fine motor skills should be evaluated in children with PAE or FASD. The high prevalence of fine motor impairment in our cohort, even in children without PAE, highlights the need for therapeutic intervention for many children in remote communities.

Pediatric hospital admissions in Indigenous children: a population-based study in remote Australia.

BACKGROUND: We analysed hospital admissions of a predominantly Aboriginal cohort of children in the remote Fitzroy Valley in Western Australia during the first 7 years of life. METHODS: All children born between January 1, 2002 and December 31, 2003 and living in the Fitzroy Valley in 2009-2010 were eligible to participate in the Lililwan Project. Of 134 eligible children, 127 (95%) completed Stage 1 (interviews of caregivers and medical record review) in 2011 and comprised our cohort. Lifetime (0-7 years) hospital admission data were available and included the dates, and reasons for admission, and comorbidities. Conditions were coded using ICD-10-AM discharge codes. RESULTS: Of the 127 children, 95.3% were Indigenous and 52.8% male. There were 314 admissions for 424 conditions in 89 (70.0%) of 127 children. The 89 children admitted had a median of five admissions (range 1-12). Hospitalization rates were similar for both genders (p = 0.4). Of the admissions, 108 (38.6%) were for 56 infants aged <12 months (median = 2.5, range = 1-8). Twelve of these admissions were in neonates (aged 0-28 days). Primary reasons for admission (0-7 years) were infections of the lower respiratory tract (27.4%), gastrointestinal system (22.7%), and upper respiratory tract (11.4%), injury (7.0%), and failure to thrive (5.4%). Comorbidities, particularly upper respiratory tract infections (18.1%), failure to thrive (13.6%), and anaemia (12.7%), were common. In infancy, primary cause for admission were infections of the lower respiratory tract (40.8%), gastrointestinal (25.9%) and upper respiratory tract (9.3%). Comorbidities included upper respiratory tract infections (33.3%), failure to thrive (18.5%) and anaemia (18.5%). CONCLUSION: In the Fitzroy Valley 70.0% of children were hospitalised at least once before age 7 years and over one third of admissions were in infants. Infections were the most common reason for admission in all age groups but comorbidities were common and may contribute to need for admission. Many hospitalizations were feasibly preventable. High admission rates reflect disadvantage, remote location and limited access to primary healthcare and outpatient services. Ongoing public health prevention initiatives including breast feeding, vaccination, healthy diet, hygiene and housing improvements are crucial, as is training of Aboriginal Health Workers to increase services in remote communities.

The Marulu Strategy 2008-2012: overcoming Fetal Alcohol Spectrum Disorder (FASD) in the Fitzroy Valley.

OBJECTIVE: Aboriginal leaders concerned about high rates of Fetal Alcohol Spectrum Disorder (FASD) in the Fitzroy Valley, remote north-western Australia, introduced restrictions on access to take-away full-strength alcohol. Following this, Aboriginal leaders engaged strategic partners in a broader strategy to address FASD in the region. The aim of this study was to develop and implement a community-led, researcher-supported, FASD strategy. METHODS: A review of literature focusing on community-led FASD strategies identified key components that informed the Marulu FASD strategy. These included strategy ownership, leadership, and governance by participating communities, and a research framework. RESULTS: Community meetings and workshops led to the development of The Marulu FASD Strategy (2008). Feasibility and community consent to conduct a FASD prevalence study (the Lililwan Project) was confirmed, and implementation was progressed (2010-2013). Concurrent FASD prevention activities were conducted. In 2012, the Marulu FASD Unit was established within a local Aboriginal organisation to sustain and coordinate ongoing strategy activities. CONCLUSIONS: Community control of public health initiatives can be achieved when Aboriginal communities prioritise issues of significant concern, and engage strategic partners to overcome them. Implications for public health: The Marulu Strategy forms a template for action to address FASD and other public health issues in Aboriginal communities in Australia and internationally.

Prevalence and profile of Neurodevelopment and Fetal Alcohol Spectrum Disorder (FASD) amongst Australian Aboriginal children living in remote communities.

BACKGROUND: Despite multiple risk factors for neurodevelopmental vulnerability, few studies have assessed neurodevelopmental performance of Australian Aboriginal children. An important risk factor for neurodevelopmental vulnerability is prenatal alcohol exposure (PAE), which places children at risk for Fetal Alcohol Spectrum Disorder (FASD). AIMS: This study assesses neurodevelopment outcomes in a population of Australian Aboriginal children with and without PAE. METHODS AND PROCEDURES: Children born in 2002/2003, and living in the Fitzroy Valley, Western Australia between April 2010 and November 2011, were eligible (N=134). Sociodemographic and antenatal data, including PAE, were collected by interview with 127/134 (95%) consenting parents/caregivers. Maternal/child medical records were reviewed. Neurodevelopment was assessed by clinicians blinded to PAE in 108/134 (81%) children and diagnoses on the FASD spectrum were assigned. OUTCOMES AND RESULTS: Neurodevelopmental disorder was documented in 34/108 children (314.8 per 1000). Any diagnosis on the FASD spectrum was made in 21/108 (194.4 per 1000) children (95% CI=131.0-279.0). CONCLUSIONS AND IMPLICATIONS: Neurodevelopmental impairment with or without PAE is highly prevalent among children in the Fitzroy Valley. Rates of diagnoses on the FASD spectrum are among the highest worldwide. Early intervention services are needed to support developmentally vulnerable children in remote communities.

Graphomotor skills in children with prenatal alcohol exposure and fetal alcohol spectrum disorder: A population-based study in remote Australia.

BACKGROUND/AIM: Few studies have examined graphomotor skills in children with prenatal alcohol exposure (PAE) or fetal alcohol spectrum disorder (FASD). METHODS: Graphomotor skills were assessed in 108 predominantly Australian Aboriginal children aged 7.5-9.6 years in remote Western Australia using clinical observations (pencil grasp; writing pressure) and standardised assessment tools (the Evaluation Tool of Children's Handwriting; and the Miller Function and Participation Scales - The Draw-a-Kid Game). Skills were compared between children (i) without PAE, (ii) PAE but not FASD and (iii) FASD. RESULTS: Most children used a transitional pencil grasp and exerted heavy handwriting pressure (83.3% and 30.6% of the cohort). The percentage of letters (M = 62.9%) and words (M = 73.3%) written legibly was low. Children with FASD were more likely than children without PAE to use a cross-thumb grasp (P = 0.027), apply heavy writing pressure (P = 0.036), be unable to write a sentence (P = 0.041) and show poorer word legibility (P = 0.041). There were no significant differences between groups for drawing outcomes, although some children with FASD drew pictures that appeared delayed for their age. There were no significant differences between children without PAE and those with PAE but who were not diagnosed with FASD. CONCLUSIONS: Overall, graphomotor skills were poor in this cohort, but children with FASD performed significantly worse than children without PAE. Findings suggest the need for improved occupational therapy services for children in remote regions and evaluation of graphomotor skills in children with PAE.

Gross motor performance in children prenatally exposed to alcohol and living in remote Australia.

AIM: This study aimed to determine the gross motor (GM) performance of Aboriginal children living in remote Australia. The relationship between GM skills, prenatal alcohol exposure (PAE) and fetal alcohol spectrum disorders (FASD) was explored. METHODS: A population-based observation study was conducted in 2011 to assess motor performance in children living in the Fitzroy Valley, Western Australia, using the Bruininks-Oseretsky Test of Motor Proficiency (BOT-2). BOT-2 data were retrospectively analysed using recently developed software enabling separation of fine and GM outcomes. RESULTS: A total of 108 children (98.1% Aboriginal; 53% male, mean age: 8.7 years) were assessed. Half (52.2%) were exposed to at least 'risky' levels of PAE, and 21 (19%) were diagnosed with an FASD. The mean GM composite score of the cohort (47.0 ± 8.4) approached the BOT-2 normative mean (50.0 ± 10) and was similar between children with and without PAE (P = 0.27). This mean score, however, was significantly lower in children with FASD than without (mean difference: -5.5 ± 20.6; P = 0.006). Compared with children without FASD, children with FASD had significant impairment in subtests for running speed and agility (mean difference ± standard deviation (SD): -2.4 ± 8.1; P = 0.003) and strength (mean difference ± SD:-2.8 ± 9.9; P = 0.004) and (ii) a higher proportion than expected had overall GM impairment (≤2 SD: 9.5%; ≤1 SD: 23.8%). In groups with PAE, no PAE and no FASD, GM function approached expected population norms. CONCLUSIONS: A higher than expected proportion of children with FASD had GM scores that indicated impairment and need for therapy. Evaluation of GM performance should routinely be included in FASD assessment to determine strategies to optimise child development.

Study protocol for screening and diagnosis of fetal alcohol spectrum disorders (FASD) among young people sentenced to detention in Western Australia.

INTRODUCTION: Prenatal alcohol exposure can cause lifelong disability, including physical, cognitive and behavioural deficits, known as fetal alcohol spectrum disorders (FASD). Among individuals with FASD, engagement with justice services is common. Little is known about the prevalence of FASD among young people engaged with the Australian justice system. This study aims to establish FASD prevalence among sentenced young people in detention in Western Australia (WA), and use the findings to develop a screening tool for use among young people entering detention. Translation of these results will guide the management and support of young people in detention and will have significant implications on the lives of young people with FASD and the future of Australian youth justice services. METHODS AND ANALYSIS: Any sentenced young person in WA aged 10-17 years 11 months is eligible to participate. Young people are assessed for FASD by a multidisciplinary team. Standardised assessment tools refined for the Australian context are used, acknowledging the language and social complexities involved. Australian diagnostic guidelines for FASD will be applied. Information is obtained from young people, responsible adults, teachers and custodial officers. Individualised results and management plans for each young person are communicated to the young person and responsible adult. Prevalence of FASD will be reported and multivariate methods used to identify variables most predictive of FASD and to optimise the predictive value of screening. ETHICS AND DISSEMINATION: Approvals have been granted by the WA Aboriginal Health Ethics Committee, University of WA Human Research Ethics Committee, Department of Corrective Services, and Department for Child Protection and Family Support. Anonymised findings will be disseminated through peer-reviewed manuscripts, presentations and the media. Extensive consultation with stakeholders (including government agencies, detention centre staff, community service providers, the young people and their families or carers) will be ongoing until findings are disseminated and translated.

Visual-motor integration, visual perception, and fine motor coordination in a population of children with high levels of Fetal Alcohol Spectrum Disorder.

BACKGROUND: Visual-motor integration (VMI) skills are essential for successful academic performance, but to date no studies have assessed these skills in a population-based cohort of Australian Aboriginal children who, like many children in other remote, disadvantaged communities, consistently underperform academically. Furthermore, many children in remote areas of Australia have prenatal alcohol exposure (PAE) and Fetal Alcohol Spectrum Disorder (FASD), which are often associated with VMI deficits. METHODS: VMI, visual perception, and fine motor coordination were assessed using The Beery-Buktenica Developmental Test of Visual-Motor Integration, including its associated subtests of Visual Perception and Fine Motor Coordination, in a cohort of predominantly Australian Aboriginal children (7.5-9.6 years, n=108) in remote Western Australia to explore whether PAE adversely affected test performance. Cohort results were reported, and comparisons made between children i) without PAE; ii) with PAE (no FASD); and iii) FASD. The prevalence of moderate (≤16th percentile) and severe (≤2nd percentile) impairment was established. RESULTS: Mean VMI scores were 'below average' (M=87.8±9.6), and visual perception scores were 'average' (M=97.6±12.5), with no differences between groups. Few children had severe VMI impairment (1.9%), but moderate impairment rates were high (47.2%). Children with FASD had significantly lower fine motor coordination scores and higher moderate impairment rates (M=87.9±12.5; 66.7%) than children without PAE (M=95.1±10.7; 23.3%) and PAE (no FASD) (M=96.1±10.9; 15.4%). CONCLUSIONS: Aboriginal children living in remote Western Australia have poor VMI skills regardless of PAE or FASD. Children with FASD additionally had fine motor coordination problems. VMI and fine motor coordination should be assessed in children with PAE, and included in FASD diagnostic assessments.

Soft neurological signs and prenatal alcohol exposure: a population-based study in remote Australia.

AIM: To identify soft neurological signs (SNS) in a population-based study of children living in remote Aboriginal communities in the Fitzroy Valley, Western Australia, born between 2002 and 2003 and explore the relationship between SNS, prenatal alcohol exposure (PAE), and fetal alcohol spectrum disorders (FASD). METHOD: The presence of SNS was assessed using the Quick Neurological Screening Test, 2nd edition (QNST-2), which has a total maximum score of 140. Higher scores indicated more SNS. 'Severe discrepancy' was defined as scores less than or equal to the fifth centile while 'moderate discrepancy' represented scores from the sixth to the 24th centile. Children were assigned FASD diagnoses using modified Canadian FASD diagnostic guidelines. RESULTS: A total of 108 of 134 (80.6%) eligible children (mean age 8y 9mo, SD=6mo, 53% male) were assessed. The median QNST-2 Total Score for all participants was within the normal category (19.0, range 4-66). However, the median QNST-2 Total Score was higher in children with than without (1) PAE (r=0.2, p=0.045) and (2) FASD (r=0.3, p=0.004). Half (8/16) of children scoring 'moderate discrepancy' and all (2/2) children scoring 'severe discrepancy' had at least three domains of central nervous system impairment. INTERPRETATION: SNS were more common in children with PAE or FASD, consistent with the known neurotoxic effect of PAE. The QNST-2 is a useful screen for subtle neurological dysfunction indicating the need for more comprehensive assessment in children with PAE or FASD.

Impairment of motor skills in children with fetal alcohol spectrum disorders in remote Australia: The Lililwan Project.

INTRODUCTION AND AIMS: We aimed to characterise motor performance in predominantly Aboriginal children living in very remote Australia, where rates of prenatal alcohol exposure (PAE) are high. Motor performance was assessed, and the relationship between motor skills, fetal alcohol spectrum disorders (FASD) and PAE was explored. DESIGN AND METHODS: Motor performance was assessed using the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition Complete Form, in a population-based study of children born in 2002 or 2003 living in the Fitzroy Valley, Western Australia. Composite scores ≥2SD (2nd percentile) and ≥1SD (16th percentile) below the mean were used respectively for FASD diagnosis and referral for treatment. FASD diagnoses were assigned using modified Canadian Guidelines. RESULTS: A total of 108 children (Aboriginal: 98.1%; male: 53%) with a mean age of 8.7 years was assessed. The cohort's mean total motor composite score (mean ± SD 47.2 ± 7.6) approached the Bruininks-Oseretsky Test of Motor Proficiency-Second Edition normative mean (50 ± 10). Motor performance was lower in children with FASD diagnosis than without (mean difference (MD) ± SD: -5.0 ± 1.8; confidence interval: -8.6 to -1.5). There was no difference between children with PAE than without (MD ± SE: -2.2 ± 1.5; confidence interval: -5.1 to 0.80). The prevalence of motor impairment (≥-2SD) was 1.9% in the entire cohort, 9.5% in children with FASD, 3.3% in children with PAE and 0.0% both in children without PAE or FASD. DISCUSSION AND CONCLUSIONS: Almost of 10% of children with FASD has significant motor impairment. Evaluation of motor function should routinely be included in assessments for FASD, to document impairment and enable targeted early intervention.[Lucas BR, Doney R, Latimer J, Watkins RE, Tsang TW, Hawkes G, Fitzpatrick JP, Oscar J, Carter M, Elliott EJ. Impairment of motor skills in children with fetal alcohol spectrum disorders in remote Australia: The Lililwan Project. Drug Alcohol Rev 2016;35:719-727].

Prevalence and patterns of alcohol use in pregnancy in remote Western Australian communities: The Lililwan Project.

INTRODUCTION AND AIMS: Alcohol use in pregnancy is thought to be common in remote Australian communities, but no population-based data are available. Aboriginal leaders in remote Western Australia invited researchers to determine the prevalence and patterns of alcohol use in pregnancy within their communities. DESIGN AND METHODS: A population-based survey of caregivers of all children born in 2002/2003 and living in the Fitzroy Valley in 2010/2011 (n = 134). Alcohol use risk was categorised using the Alcohol Use Disorders Identification Test consumption subset (AUDIT-C) tool. Birth and child outcomes were determined by interview, medical record review and physical examination. RESULTS: 127/134 (95%) eligible caregivers participated: 78% were birth mothers, 95% were Aboriginal and 55% reported alcohol use in index pregnancies; 88% reported first trimester drinking and 53% drinking in all trimesters. AUDIT-C scores were calculated for 115/127 women, of whom 60 (52%) reported alcohol use in pregnancy. Of the 60 women who drank (AUDIT-C score ≥ 1), 12% drank daily/almost daily, 33% drank 2-3 times per week; 71% drank ≥ 10 standard drinks on a typical occasion; 95% drank at risky or high-risk levels (AUDIT-C score ≥ 4). Mean AUDIT-C score was 8.5 ± 2.3 (range 2-12). The most common drinking pattern was consumption of ≥ 10 standard drinks either 2-4 times per month (27%) or 2-3 times per week (27%). DISCUSSION AND CONCLUSIONS: High-risk alcohol use in pregnancy is common in remote, predominantly Aboriginal communities in north western Australia. Prevention strategies to reduce prenatal alcohol use are urgently needed.

Prevalence of fetal alcohol syndrome in a population-based sample of children living in remote Australia: the Lililwan Project.

AIM: Aboriginal leaders concerned about high rates of alcohol use in pregnancy invited researchers to determine the prevalence of fetal alcohol syndrome (FAS) and partial fetal alcohol syndrome (pFAS) in their communities. METHODS: Population-based prevalence study using active case ascertainment in children born in 2002/2003 and living in the Fitzroy Valley, in Western Australia (April 2010-November 2011) (n = 134). Socio-demographic and antenatal data, including alcohol use in pregnancy, were collected by interview with 127/134 (95%) consenting parents/care givers. Maternal/child medical records were reviewed. Interdisciplinary assessments were conducted for 108/134 (81%) children. FAS/pFAS prevalence was determined using modified Canadian diagnostic guidelines. RESULTS: In 127 pregnancies, alcohol was used in 55%. FAS or pFAS was diagnosed in 13/108 children, a prevalence of 120 per 1000 (95% confidence interval 70-196). Prenatal alcohol exposure was confirmed for all children with FAS/pFAS, 80% in the first trimester and 50% throughout pregnancy. Ten of 13 mothers had Alcohol Use Disorders Identification Test scores and all drank at a high-risk level. Of children with FAS/pFAS, 69% had microcephaly, 85% had weight deficiency and all had facial dysmorphology and central nervous system abnormality/impairment in three to eight domains. CONCLUSIONS: The population prevalence of FAS/pFAS in remote Aboriginal communities of the Fitzroy Valley is the highest reported in Australia and similar to that reported in high-risk populations internationally. Results are likely to be generalisable to other age groups in the Fitzroy Valley and other remote Australian communities with high-risk alcohol use during pregnancy. Prevention of FAS/pFAS is an urgent public health challenge.