RESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal/epidemiologia , Havaiano Nativo ou Outro Ilhéu do Pacífico , Classe Social , População Branca , Desempenho Acadêmico , Adaptação Psicológica , Adolescente , Adulto , Atenção , Transtorno do Deficit de Atenção com Hiperatividade/epidemiologia , Transtorno do Deficit de Atenção com Hiperatividade/psicologia , Criança , Pré-Escolar , Estudos Transversais , Função Executiva , Feminino , Transtornos do Espectro Alcoólico Fetal/etnologia , Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Transtornos do Espectro Alcoólico Fetal/psicologia , Humanos , Idioma , Masculino , Northern Territory/epidemiologia , Estudos Retrospectivos , População Rural , Distribuição por Sexo , Transtornos do Sono-Vigília/epidemiologia , Transtornos do Sono-Vigília/fisiopatologia , Austrália Ocidental/epidemiologia , Adulto JovemRESUMO
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.
Assuntos
Saúde da Criança/etnologia , Disparidades nos Níveis de Saúde , Hospitalização/estatística & dados numéricos , Havaiano Nativo ou Outro Ilhéu do Pacífico , Saúde da População Rural/etnologia , Criança , Pré-Escolar , Comorbidade , Feminino , Acessibilidade aos Serviços de Saúde , Humanos , Lactente , Recém-Nascido , Masculino , Estudos Retrospectivos , Austrália Ocidental/epidemiologiaRESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Transtornos das Habilidades Motoras/etiologia , Destreza Motora , Efeitos Tardios da Exposição Pré-Natal/fisiopatologia , Criança , Estudos de Coortes , Feminino , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Transtornos do Espectro Alcoólico Fetal/epidemiologia , Humanos , Masculino , Transtornos das Habilidades Motoras/diagnóstico , Transtornos das Habilidades Motoras/epidemiologia , Testes Neuropsicológicos , Gravidez , Efeitos Tardios da Exposição Pré-Natal/diagnóstico , Efeitos Tardios da Exposição Pré-Natal/epidemiologia , Efeitos Tardios da Exposição Pré-Natal/etiologia , Prevalência , Desempenho Psicomotor , Austrália Ocidental/epidemiologiaRESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Escrita Manual , Destreza Motora/fisiologia , Havaiano Nativo ou Outro Ilhéu do Pacífico , Efeitos Tardios da Exposição Pré-Natal/fisiopatologia , Criança , Feminino , Transtornos do Espectro Alcoólico Fetal/etnologia , Humanos , Gravidez , Efeitos Tardios da Exposição Pré-Natal/etnologia , Austrália Ocidental/epidemiologiaRESUMO
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.
Assuntos
Doenças do Sistema Nervoso/etiologia , Efeitos Tardios da Exposição Pré-Natal/epidemiologia , Efeitos Tardios da Exposição Pré-Natal/fisiopatologia , Austrália/epidemiologia , Criança , Estudos de Coortes , Planejamento em Saúde Comunitária , Teste de Esforço , Feminino , Transtornos do Espectro Alcoólico Fetal/epidemiologia , Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Humanos , Masculino , Havaiano Nativo ou Outro Ilhéu do Pacífico , Doenças do Sistema Nervoso/diagnóstico , Exame Neurológico , Avaliação de Resultados em Cuidados de Saúde , Gravidez , Efeitos Tardios da Exposição Pré-Natal/etnologia , Estatísticas não ParamétricasRESUMO
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.
Assuntos
Desenvolvimento Infantil/efeitos dos fármacos , Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Destreza Motora/efeitos dos fármacos , Efeitos Tardios da Exposição Pré-Natal/fisiopatologia , Criança , Testes Diagnósticos de Rotina , Feminino , Humanos , Masculino , Havaiano Nativo ou Outro Ilhéu do Pacífico , Gravidez , Estudos Retrospectivos , População RuralRESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal/etnologia , Havaiano Nativo ou Outro Ilhéu do Pacífico/estatística & dados numéricos , Saúde da População Rural/etnologia , Adolescente , Adulto , Consumo de Bebidas Alcoólicas/efeitos adversos , Consumo de Bebidas Alcoólicas/etnologia , Criança , Feminino , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Transtornos do Espectro Alcoólico Fetal/etiologia , Humanos , Masculino , Comportamento Materno/etnologia , Gravidez , Prevalência , Fatores de Risco , Saúde da População Rural/estatística & dados numéricos , Austrália Ocidental/epidemiologia , Adulto JovemRESUMO
BACKGROUND: Fetal alcohol spectrum disorder (FASD) is known to be under-recognised in Australia. The use of standard methods to identify when to refer individuals who may have FASD for specialist assessment could help improve the identification of this disorder. The purpose of this study was to develop referral criteria for use in Australia. METHOD: An online survey about FASD screening and diagnosis in Australia, which included 23 statements describing criteria for referral for fetal alcohol syndrome (FAS) and FASD based on published recommendations for referral in North America, was sent to 139 health professionals who had expertise or involvement in FASD screening or diagnosis. Survey findings and published criteria for referral were subsequently reviewed by a panel of 14 investigators at a consensus development workshop where criteria for referral were developed. RESULTS: Among the 139 health professionals who were sent the survey, 103 (74%) responded, and 90 (65%) responded to the statements on criteria for referral. Over 80% of respondents agreed that referral for specialist evaluation should occur when there is evidence of significant prenatal alcohol exposure, defined as 7 or more standard drinks per week and at least 3 standard drinks on any one day, and more than 70% agreed with 13 of the 16 statements that described criteria for referral other than prenatal alcohol exposure. Workshop participants recommended five independent criteria for referral: confirmed significant prenatal alcohol exposure; microcephaly and confirmed prenatal alcohol exposure; 2 or more significant central nervous system (CNS) abnormalities and confirmed prenatal alcohol exposure; 3 characteristic FAS facial anomalies; and 1 characteristic FAS facial anomaly, growth deficit and 1 or more CNS abnormalities. CONCLUSION: Referral criteria recommended for use in Australia are similar to those recommended in North America. There is a need to develop resources to raise awareness of these criteria among health professionals and evaluate their feasibility, acceptability and capacity to improve the identification of FASD in Australia.
Assuntos
Atitude do Pessoal de Saúde , Consenso , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Encaminhamento e Consulta/normas , Consumo de Bebidas Alcoólicas/efeitos adversos , Austrália , Feminino , Transtornos do Espectro Alcoólico Fetal/etiologia , Pesquisas sobre Atenção à Saúde , Humanos , Masculino , Comportamento Materno , Gravidez , Efeitos Tardios da Exposição Pré-Natal/diagnóstico , Efeitos Tardios da Exposição Pré-Natal/etiologia , Fatores de RiscoRESUMO
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.
Assuntos
Metilação de DNA , Efeitos Tardios da Exposição Pré-Natal , Animais , Metilação de DNA/efeitos dos fármacos , Feminino , Gravidez , Efeitos Tardios da Exposição Pré-Natal/genética , Camundongos , Humanos , Dieta , Masculino , Etanol/efeitos adversos , Etanol/toxicidade , Camundongos Endogâmicos C57BL , Modelos Animais de Doenças , Encéfalo/efeitos dos fármacos , Encéfalo/embriologia , Encéfalo/metabolismo , Transtornos do Espectro Alcoólico Fetal/genética , Fígado/efeitos dos fármacos , Fígado/metabolismo , Fígado/embriologiaRESUMO
BACKGROUND: The Lililwan Project is the first population-based study to determine Fetal Alcohol Spectrum Disorders (FASD) prevalence in Australia and was conducted in the remote Fitzroy Valley in North Western Australia. The diagnostic process for FASD requires accurate assessment of gross and fine motor functioning using standardised cut-offs for impairment. The Bruininks-Oseretsky Test of Motor Proficiency, Second Edition (BOT-2) is a norm-referenced assessment of motor function used worldwide and in FASD clinics in North America. It is available in a Complete Form with 53 items or a Short Form with 14 items. Its reliability in measuring motor performance in children exposed to alcohol in utero or living in remote Australian Aboriginal communities is unknown. METHODS: A prospective inter-rater and test-retest reliability study was conducted using the BOT-2 Short Form. A convenience sample of children (n = 30) aged 7 to 9 years participating in the Lililwan Project cohort (n = 108) study, completed the reliability study. Over 50% of mothers of Lililwan Project children drank alcohol during pregnancy. Two raters simultaneously scoring each child determined inter-rater reliability. Test-retest reliability was determined by assessing each child on a second occasion using predominantly the same rater. Reliability was analysed by calculating Intra-Class correlation Coefficients, ICC(2,1), Percentage Exact Agreement (PEA) and Percentage Close Agreement (PCA) and measures of Minimal Detectable Change (MDC) were calculated. RESULTS: Thirty Aboriginal children (18 male, 12 female: mean age 8.8 years) were assessed at eight remote Fitzroy Valley communities. The inter-rater reliability for the BOT-2 Short Form score sheet outcomes ranged from 0.88 (95%CI, 0.77 - 0.94) to 0.92 (95%CI, 0.84 - 0.96) indicating excellent reliability. The test-retest reliability (median interval between tests being 45.5 days) for the BOT-2 Short Form score sheet outcomes ranged from 0.62 (95%CI, 0.34 - 0.80) to 0.73 (95%CI, 0.50 - 0.86) indicating fair to good reliability. The raw score MDC was 6.12. CONCLUSION: The BOT-2 Short Form has acceptable reliability for use in remote Australian Aboriginal communities and will be useful in determining motor deficits in children exposed to alcohol prenatally. This is the first known study evaluating the reliability of the BOT-2 Short Form, either in the context of assessment for FASD or in Aboriginal children.
Assuntos
Transtornos do Espectro Alcoólico Fetal/diagnóstico , Destreza Motora/fisiologia , Havaiano Nativo ou Outro Ilhéu do Pacífico , Aptidão Física/fisiologia , Efeitos Tardios da Exposição Pré-Natal/diagnóstico , Desempenho Psicomotor/fisiologia , Criança , Feminino , Transtornos do Espectro Alcoólico Fetal/fisiopatologia , Humanos , Masculino , Testes Neuropsicológicos , Gravidez , Efeitos Tardios da Exposição Pré-Natal/fisiopatologia , Estudos Prospectivos , Reprodutibilidade dos Testes , Austrália OcidentalRESUMO
BACKGROUND: A battery of clinical assessments was used in the Lililwan Project, Australia's first population-based Fetal Alcohol Spectrum Disorders (FASD) prevalence study, conducted in the remote Fitzroy Valley, Western Australia. One objective was to develop and assess test-retest reliability of an acceptable questionnaire for collecting health information in remote Aboriginal communities feasible for use in the Lililwan Project. METHODS: A questionnaire was developed by paediatricians to assist in diagnosis of FASD. Content was based on a literature review of FASD diagnostic criteria, existing questionnaires and risk factors for FASD and birth defects. Aboriginal community members, including qualified Aboriginal language interpreters, adapted the questionnaire to ensure language and cultural components were appropriate for use in the Fitzroy Valley. Locally developed pictorial aids were used for gathering accurate information on alcohol use. Aboriginal 'community navigators' assisted researchers to translate the questions into Kimberley Kriol or local Aboriginal languages depending on participant preference. RESULTS: The full questionnaire contained 112 items and took 50 minutes to administer. For a subset of 14 items from the full questionnaire percent exact agreement between raters ranged from 59-100%, and was below 70% for only 1 question. Test-retest reliability was excellent (Kappa 0.81-1.00) for 5 items, substantial (Kappa 0.61-0.80) for 5 items, and moderate, fair or slight (Kappa ≤0.60) for the remaining 4 items tested. Test-retest reliability for questions relating to alcohol use in pregnancy was excellent. When questions had moderate, fair or slight agreement, information was obtained from alternate sources e.g. medical records. Qualitative feedback from parents/carers confirmed acceptability of the questionnaire. CONCLUSIONS: This questionnaire had acceptable test-retest reliability and could be used to collect demographic, socio-cultural and biomedical information relevant to the diagnosis of FASD in Aboriginal communities throughout Australia and elsewhere. Community input is crucial when developing and administering questionnaires for use in cross-cultural contexts.
Assuntos
Consumo de Bebidas Alcoólicas/epidemiologia , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Inquéritos e Questionários , Feminino , Transtornos do Espectro Alcoólico Fetal/epidemiologia , Humanos , Projetos Piloto , Gravidez , Reprodutibilidade dos Testes , Fatores de Risco , Austrália Ocidental/epidemiologiaRESUMO
BACKGROUND: Fetal alcohol spectrum disorders (FASD) are underdiagnosed in Australia, and health professionals have endorsed the need for national guidelines for diagnosis. The aim of this study was to develop consensus recommendations for the diagnosis of FASD in Australia. METHODS: A panel of 13 health professionals, researchers, and consumer and community representatives with relevant expertise attended a 2-day consensus development workshop to review evidence on the screening and diagnosis of FASD obtained from a systematic literature review, a national survey of health professionals and community group discussions. The nominal group technique and facilitated discussion were used to review the evidence on screening and diagnosis, and to develop consensus recommendations for the diagnosis of FASD in Australia. RESULTS: The use of population-based screening for FASD was not recommended. However, there was consensus support for the development of standard criteria for referral for specialist diagnostic assessment. Participants developed consensus recommendations for diagnostic categories, criteria and assessment methods, based on the adaption of elements from both the University of Washington 4-Digit Diagnostic Code and the Canadian guidelines for FASD diagnosis. Panel members also recommended the development of resources to: facilitate consistency in referral and diagnostic practices, including comprehensive clinical guidelines and assessment instruments; and to support individuals undergoing assessment and their parents or carers. CONCLUSIONS: These consensus recommendations provide a foundation for the development of guidelines and other resources to promote consistency in the diagnosis of FASD in Australia. Guidelines for diagnosis will require review and evaluation in the Australian context prior to national implementation as well as periodic review to incorporate new knowledge.
Assuntos
Transtornos do Espectro Alcoólico Fetal/diagnóstico , Guias de Prática Clínica como Assunto , Austrália , Medicina Baseada em Evidências , Feminino , Humanos , Recém-Nascido , Masculino , Programas de RastreamentoRESUMO
BACKGROUND: There is little reliable information on the prevalence of fetal alcohol spectrum disorders (FASD) in Australia and no coordinated national approach to facilitate case detection. The aim of this study was to identify health professionals' perceptions about screening for FASD in Australia. METHOD: A modified Delphi process was used to assess perceptions of the need for, and the process of, screening for FASD in Australia. We recruited a panel of 130 Australian health professionals with experience or expertise in FASD screening or diagnosis. A systematic review of the literature was used to develop Likert statements on screening coverage, components and assessment methods which were administered using an online survey over two survey rounds. RESULTS: Of the panel members surveyed, 95 (73%) responded to the questions on screening in the first survey round and, of these, 81 (85%) responded to the second round. Following two rounds there was consensus agreement on the need for targeted screening at birth (76%) and in childhood (84%). Participants did not reach consensus agreement on the need for universal screening at birth (55%) or in childhood (40%). Support for targeted screening was linked to perceived constraints on service provision and the need to examine the performance, costs and benefits of screening.For targeted screening of high risk groups, we found highest agreement for siblings of known cases of FASD (96%) and children of mothers attending alcohol treatment services (93%). Participants agreed that screening for FASD primarily requires assessment of prenatal alcohol exposure at birth (86%) and in childhood (88%), and that a checklist is needed to identify the components of screening and criteria for referral at birth (84%) and in childhood (90%). CONCLUSIONS: There is an agreed need for targeted but not universal screening for FASD in Australia, and sufficient consensus among health professionals to warrant development and evaluation of standardised methods for targeted screening and referral in the Australian context. Participants emphasised the need for locally-appropriate, evidence-based approaches to facilitate case detection, and the importance of ensuring that screening and referral programs are supported by adequate diagnostic and management capacity.
Assuntos
Atitude do Pessoal de Saúde , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Programas de Rastreamento , Austrália , Técnica Delphi , Feminino , Humanos , Masculino , Programas de Rastreamento/métodos , Programas de Rastreamento/organização & administração , Gravidez , Inquéritos e QuestionáriosRESUMO
BACKGROUND: Australia's commitment to consumer and community participation in health and medical research has grown over the past decade. Participatory research models of engagement are the most empowering for consumers. METHODS: As part of a project to develop a diagnostic instrument for fetal alcohol spectrum disorders (FASD) in Australia (FASD Project), the Australian FASD Collaboration (Collaboration), including a consumer advocate and two consumer representatives, was established. On completion of the FASD Project an on-line survey of Collaboration members was conducted to assess their views on consumer involvement. Women in the community were also invited to participate in Community Conversations to discuss real life situations regarding communications with health professionals about alcohol and pregnancy. Community Conversation feedback was analysed qualitatively and attendees were surveyed about their views of the Community Conversation process. RESULTS: The on-line survey was completed by 12 members of the Collaboration (71%). Consumer and community participation was considered important and essential, worked well, and was integral to the success of the project. The 32 women attending the Community Conversations generated 500 statements that made reference to prevention, how information and messages are delivered, and appropriate support for women. Nearly all the attendees at the Community Conversations (93%) believed that they had an opportunity to put forward their ideas and 96% viewed the Community Conversations as a positive experience. CONCLUSIONS: The successful involvement of consumers and the community in the FASD Project can be attributed to active consumer and community participation, which included continued involvement throughout the project, funding of participation activities, and an understanding of the various contributions by the Collaboration members.
Assuntos
Participação da Comunidade , Transtornos do Espectro Alcoólico Fetal/diagnóstico , Diagnóstico Pré-Natal , Austrália , Consenso , Comportamento Cooperativo , Feminino , Humanos , GravidezRESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal , Feminino , Gravidez , Humanos , Transtornos do Espectro Alcoólico Fetal/diagnóstico , AustráliaRESUMO
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.
Assuntos
Transtornos do Espectro Alcoólico Fetal , Humanos , Feminino , Gravidez , Idoso , Acomodação Ocular , Bases de Dados Factuais , Eletrônica , EmpregoRESUMO
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.
Assuntos
Consumo de Bebidas Alcoólicas/epidemiologia , Transtornos do Espectro Alcoólico Fetal/prevenção & controle , Havaiano Nativo ou Outro Ilhéu do Pacífico/estatística & dados numéricos , Efeitos Tardios da Exposição Pré-Natal/prevenção & controle , Adulto , Consumo de Bebidas Alcoólicas/efeitos adversos , Consumo de Bebidas Alcoólicas/etnologia , Austrália/epidemiologia , Feminino , Transtornos do Espectro Alcoólico Fetal/etnologia , Humanos , Gravidez , Primeiro Trimestre da Gravidez , Terceiro Trimestre da Gravidez , Efeitos Tardios da Exposição Pré-Natal/etnologia , Prevalência , Características de ResidênciaRESUMO
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.
Assuntos
Função Executiva/fisiologia , Serviços de Saúde do Indígena , Havaiano Nativo ou Outro Ilhéu do Pacífico , Serviços de Saúde Escolar , Instituições Acadêmicas , Autocontrole , Estudantes , Austrália , Criança , Pré-Escolar , Docentes , Feminino , Humanos , Masculino , Pais , Inquéritos e QuestionáriosRESUMO
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.