Diagnostic Accuracy and economic value of a Tiered Assessment for Fetal Alcohol Spectrum Disorder (DATAforFASD): Protocol.

INTRODUCTION: Australian practices for diagnosing fetal alcohol spectrum disorder (FASD) are lengthy and require specialist expertise. Specialist teams are based in urban locations; they are expensive and have prolonged waitlists. Innovative, flexible solutions are needed to ensure First Nations children living in rural/remote communities have culturally appropriate and equitable access to timely diagnosis and support. This study compares the accuracy of rapid assessments (index tests) that can be administered by a range of primary healthcare practitioners to specialist standardised FASD assessments (reference tests). The cost-efficiency of index tests will be compared with reference tests. METHODS AND ANALYSIS: At least 200 children aged 6-16 years at-risk of FASD will be recruited across at least seven study sites. Following standards for reporting diagnostic accuracy study (STARD) guidelines, all children will complete index and reference tests. Diagnostic accuracy statistics (including receiver operating curves, sensitivity, specificity, positive and negative predictive values and likelihood ratios) will identify whether rapid assessments can accurately identify: (1) the presence of an FASD diagnosis and (2) impairment in each neurodevelopmental domain, compared to comprehensive assessments. Direct and indirect healthcare costs for index tests compared to reference tests will be collected in primary healthcare and specialist settings. ETHICS AND DISSEMINATION OF RESULTS: Children's Health Queensland Hospital and Health Service Human Research Ethics Committee (HREC/20/QCHQ/63173); Griffith University Human Research Ethics Committee (2020/743). Results will assist in validating the use of index tests as part of a tiered neurodevelopmental assessment process that was co-designed with First Nations community and primary healthcare practitioners. Outcomes will be summarised and provided to participating practitioners and sites, and disseminated to community health services and consumers. Findings will be presented at national and international conferences and published in peer-reviewed journals. TRIAL REGISTRATION NUMBER: ACTRN12622000498796.

Rates of early intervention services in children born extremely preterm/extremely low birthweight.

AIM: To determine the rates of early intervention (EI) service use in extremely preterm (EP, <28 weeks' gestation) or extremely low birthweight (ELBW, <1000 g) infants between 1991 and 2013, and identify biological or socio-economic factors associated with receiving EI. METHODS: Participants comprised consecutive EP or ELBW survivors born in 1991-1992, 1997 or 2005 in Victoria, Australia, and randomly selected, matched term-born controls. The main outcome measure was parent-reported EI participation up to 8 years of age. Neurodevelopmental outcomes and socio-economic risk factors were compared with EI participation to identify associations among the preterm groups. RESULTS: The rates of EI were higher in the preterm groups than the control groups overall (odds ratio 4.29, 95% confidence interval 3.28, 5.59, P < 0.001), and the rates of EI rose significantly over time - from 42% in the 1991-1992 preterm cohort to 64% in the 2005 preterm cohort. Among the preterm groups, post-natal corticosteroid therapy, cystic periventricular leukomalacia and surgery in the newborn period were all independently associated with increased odds of receiving EI. Increased severity of disability was associated with higher rates of EI. The majority (95%) of preterm children with a physical impairment received EI, compared with only 73% of children with a cognitive impairment alone. EI participation rates were independent of social risk. CONCLUSION: EI participation is high in the EP population, and rates of EI use have increased over time. Contrary to previous reports, social risk factors were not found to be associated with EI use.