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Comments and Reflections on Ethics in Screening for Biomarkers of Prenatal Alcohol Exposure
Abstract
Early identification of and intervention for fetal alcohol spectrum disorder (FASD) has been shown to optimize outcomes for affected individuals. Detecting biomarkers of prenatal alcohol exposure (PAE) in neonates may assist in the identification of children at risk of FASD enabling targeted early interventions. Despite these potential benefits, complicated ethical issues arise in screening for biomarkers of PAE and these must be addressed prior to the implementation of screening programs. Here, we identify and comment, based on a North American perspective, on concerns raised in the current ethical, social, and legal literature related to meconium screening for PAE. Major ethical concerns revolve around the targeting of populations for PAE screening, consent and respect for persons, stigma and participation rates, the cost-benefit analysis of a screening program, consequences of false-positive and false-negative test results, confidentiality and appropriate follow-up to positive screen results, and the use of screen results for criminal prosecution. We identify gaps in the literature on screening for PAE, most notably related to a lack of stakeholder perspectives (e.g., parents, healthcare providers) about screening and the ethical challenges it presents.
... 29,39,40 Identifying Screening Goals Practitioners and organizations hoping to improve identification of FASD have considerations to review prior to implementing a specific screening tool or practice, including an overarching ethical imperative to ensure that the benefits outweigh any potential risks. 22,45,46 The primary goals and intended outcomes associated with FASD identification and screening must be established early on to help orient organizations to any internal or external changes to policies, procedures, relationships, and/or resources that must first be undertaken to ensure meaningful outcomes of screening. 41 Broader goals likely include improving identification of FASD and facilitating access to enhanced care, supports, and interventions, ultimately improving outcomes for individuals, families, and society. ...
... Indeed, dignity of the person and autonomy are key ethical considerations in developing and implementing FASD screening practices. 22,45,46 In addition, care must go into planning for meaningful and ongoing informed consent procedures, given the additional support needs that may be present for people who may have FASD. 22,46,48 This may include thoughtfully reviewing the purpose, process, and potential outcomes of FASD screening, using language aligned with an individual's capacities and needs, checking in on comprehension using active paraphrasing and other approaches, appropriate training and support is provided to staff engaged in the screening process, and practical considerations such as associated costs, administration time, and impacts to service delivery. ...
... 22,45,46 In addition, care must go into planning for meaningful and ongoing informed consent procedures, given the additional support needs that may be present for people who may have FASD. 22,46,48 This may include thoughtfully reviewing the purpose, process, and potential outcomes of FASD screening, using language aligned with an individual's capacities and needs, checking in on comprehension using active paraphrasing and other approaches, appropriate training and support is provided to staff engaged in the screening process, and practical considerations such as associated costs, administration time, and impacts to service delivery. 25,41 For organizations and settings already conducting in-depth screening for other mental health and neurodevelopmental concerns, it may be possible to adapt current practices to incorporate FASD screening (and evaluate implementation processes and outcomes) rather than adopt a novel approach, particularly in the context of time and resource constraints. ...
The need to improve identification and understanding of individuals who have fetal alcohol spectrum disorder (FASD), including their strengths and challenges, is being increasingly recognized. Identification of FASD via screening is an important system-level intervention that may serve to improve early and accurate recognition of individuals who may have FASD, facilitate the provision of appropriately tailored support and interventions, and in doing so, foster healthy and positive outcomes for individuals and families. Effective and ethical implementation of FASD screening practices requires consideration of several factors for success, ensuring that resulting benefits outweigh potential harms. Using an implementation science framework, this topical review aims to provide an overview of these key considerations in order to guide further research and support practice and decision-making for service providers, organizations, and policy makers in the implementation of FASD identification and screening practices. These include prioritizing partnerships with stakeholders; taking a person-centered and ethical approach to FASD identification and screening; applying rigorous methodological research approaches to screening tool development, validation, and evaluation; increasing broader FASD awareness and response capacity at the system level; and advocating for continued policy reform and resources to enhance effective community-based support andinterventions following identification.
... 29,39,40 Identifying Screening Goals Practitioners and organizations hoping to improve identification of FASD have considerations to review prior to implementing a specific screening tool or practice, including an overarching ethical imperative to ensure that the benefits outweigh any potential risks. 22,45,46 The primary goals and intended outcomes associated with FASD identification and screening must be established early on to help orient organizations to any internal or external changes to policies, procedures, relationships, and/or resources that must first be undertaken to ensure meaningful outcomes of screening. 41 Broader goals likely include improving identification of FASD and facilitating access to enhanced care, supports, and interventions, ultimately improving outcomes for individuals, families, and society. ...
... Indeed, dignity of the person and autonomy are key ethical considerations in developing and implementing FASD screening practices. 22,45,46 In addition, care must go into planning for meaningful and ongoing informed consent procedures, given the additional support needs that may be present for people who may have FASD. 22,46,48 This may include thoughtfully reviewing the purpose, process, and potential outcomes of FASD screening, using language aligned with an individual's capacities and needs, checking in on comprehension using active paraphrasing and other approaches, appropriate training and support is provided to staff engaged in the screening process, and practical considerations such as associated costs, administration time, and impacts to service delivery. ...
... 22,45,46 In addition, care must go into planning for meaningful and ongoing informed consent procedures, given the additional support needs that may be present for people who may have FASD. 22,46,48 This may include thoughtfully reviewing the purpose, process, and potential outcomes of FASD screening, using language aligned with an individual's capacities and needs, checking in on comprehension using active paraphrasing and other approaches, appropriate training and support is provided to staff engaged in the screening process, and practical considerations such as associated costs, administration time, and impacts to service delivery. 25,41 For organizations and settings already conducting in-depth screening for other mental health and neurodevelopmental concerns, it may be possible to adapt current practices to incorporate FASD screening (and evaluate implementation processes and outcomes) rather than adopt a novel approach, particularly in the context of time and resource constraints. ...
The need to improve identification and understanding of individuals who have fetal alcohol spectrum dis-order (FASD), including their strengths and challenges, is being increasingly recognized. Identification of FASD via screening is an important system-level intervention that may serve to improve early and accurate recognition of individuals who may have FASD, facilitate the provision of appropriately tailored sup-port and interventions, and in doing so, foster healthy and positive outcomes for individuals and families. Effective and ethical implementation of FASD screening practices requires consideration of several factors for success, ensuring that resulting benefits outweigh potential harms. Using an implementation science framework, this topical review aims to provide an overview of these key considerations in order to guide further research and support practice and decision-making for service providers, organizations, and policy makers in the implementation of FASD identification and screening practices. These include prioritizing partnerships with stakeholders; taking a person-centered and ethical approach to FASD identification and screening; applying rigorous methodological research approaches to screening tool development, validation, and evaluation; increasing broader FASD awareness and response capacity at the system level; and advocating for continued policy reform and resources to enhance effective community-based support and interventions following identification.
... 196 More research is required to establish which method is most feasible and acceptable to stakeholders including clinicians, policy makers and families. 140,186,196,197 Assay methods for the biomarkers included in this review were highly variable. This is likely to account for some of the observed heterogeneity in findings. ...
... There is no consensus on what level of diagnostic accuracy is acceptable for objective measurement of PAE. Many screening initiatives prioritise sensitivity over specificity and thus permit a high number of false positive results to maximise early detection of asymptomatic conditions.186 As early diagnosis and intervention are associated with improved outcomes for children with FASD 65 it could be argued that a test with high sensitivity could be favoured over specificity, as it may support appropriate monitoring and follow-up among children with suspected PAE.However, there are ethical considerations. ...
... As early diagnosis and intervention are associated with improved outcomes for children with FASD 65 it could be argued that a test with high sensitivity could be favoured over specificity, as it may support appropriate monitoring and follow-up among children with suspected PAE.However, there are ethical considerations. Prenatal alcohol use is an emotive issue and false positive errors may lead to stigmatisation, unnecessary burden on healthcare resources, and may even be used in legal proceedings against mothers.113,127,140,186,187 Conversely, false negative errors represent a missed opportunity to provide support to those affected by PAE.127 ...
... In addition, screening throughout pregnancy and of the neonate may allow infants affected by maternal alcohol consumption to be identified. However, screening raises ethical and legal issues related to consent, individual rights and freedoms, the rights of the unborn child and stigmatisation (Mizejewski, 2010;Zizzo et al., 2013). For some women and their partners, the situation is complicated further by confusion regarding what is a 'safe' amount of alcohol to consume in pregnancy (Anderson et al., 2014;Bearer et al., 2004;Mukherjee et al., 2005;Stade et al., 2009;Van der Wulp et al., 2013). ...
... There have been numerous studies looking at levels of alcohol exposure in pregnant women, using both biomarkers and self-report (Larsson et al., 1983;Magnusson et al., 2005;Savage et al., 2002). Nevertheless, despite the widespread use of self-report and growing evidence of the technical feasibility of biomarker analysis, there are very limited data regarding the acceptability of screening (Anderson et al., 2014;Mizejewski, 2010;Shipton et al., 2013;Zizzo et al., 2013). Our study had two aims: (a) to investigate beliefs and practice regarding alcohol consumption in pregnancy and (b) to investigate whether biomarker screening for alcohol use in pregnancy was acceptable to women and their partners attending antenatal clinics in north-east England. ...
... Furthermore, the validity of simple screening questionnaires in providing an accurate record of maternal alcohol consumption is unclear. An alternative approach is to use biomarkers, but the validity and usefulness of these tests is as yet unclear and some tests can be time-consuming and expensive (Bearer et al., 2004;Mizejewski, 2010;Van der Wulp et al., 2013;Zizzo et al., 2013). ...
Providing antenatal and postnatal support for women who drink alcohol in pregnancy is only possible if those at risk can be identified. However, screening will only be helpful if women feel comfortable with the method used. We conducted a survey of pregnant women and their partners to investigate self-reported beliefs and practice regarding drinking during pregnancy and the acceptability of screening. Pregnant women and their partners attending antenatal clinics in North-East England were asked to complete a short survey regarding their alcohol consumption in pregnancy, their beliefs about safe levels of alcohol in pregnancy and whether they would be happy to have their blood or their baby’s meconium analysed for alcohol biomarkers. The data were summarised using descriptive statistics and thematic analysis. A total of 171 pregnant women and 41 partners participated. Of the pregnant women, 153 (89.5%) felt women should abstain from alcohol consumption, although only 70 (40.9%) reported not drinking in pregnancy. Of 96 women who reported drinking in pregnancy and reported when they stopped, all but six (6.3%) stopped drinking when they found out they were pregnant. Of women and partners who recorded an answer, 177 (87.2%) said they would consent to blood biomarker analysis. Confusion over what level of alcohol is safe and using screening as an opportunity for education and support emerged as key themes from free-text responses. Most women viewed screening for alcohol in pregnancy positively, although its acceptability in the small number of women who continue to drink is unclear.
... Many screening initiatives prioritize sensitivity over specificity and thus permit a high number of by guest on September 20, 2016 Downloaded from false-positive results to maximize early detection of asymptomatic conditions. 93 Early diagnosis and intervention are associated with improved outcomes for children with FASD, 43 and thus it could be argued that a test with high sensitivity could be favored over specificity in this context because it may facilitate appropriate monitoring and follow-up among children with positive PAE screens. However, PAE is a highly emotive topic, and false-positive errors may lead to stigmatization and unnecessary burden on health care resources, and may even be used in legal proceedings against mothers. ...
... Given the implications of both types of diagnostic error, various authors have suggested that both high sensitivity and specificity are a prerequisite for the introduction of PAE screening. 55,93,96 ...
... More research is required to establish which method is most feasible and acceptable to stakeholders, including clinicians, policy makers, and families. 93,94,107,108 Assay methods for the biomarkers included in this review were highly variable. This is likely to account for some of the observed heterogeneity in findings. ...
CONTEXT: Objective measurement of prenatal alcohol exposure (PAE) is essential for identifying children at risk for adverse outcomes, including fetal alcohol spectrum disorders. Biomarkers have been advocated for use in universal screening programs, but their validity has not been comprehensively evaluated.
OBJECTIVE: To systematically review the validity of objective measures of PAE.
DATA SOURCES: Thirteen electronic databases and supplementary sources were searched for studies published between January 1990 and October 2015.
STUDY SELECTION: Eligible studies were those that evaluated the diagnostic accuracy of objective measures of PAE.
DATA EXTRACTION: Three reviewers independently verified study inclusion, quality assessments,
and extracted data.
RESULTS: Twelve studies met inclusion criteria. Test performance varied widely across studies of maternal blood (4 studies; sensitivity 0%–100%, specificity 79%–100%), maternal hair (2 studies; sensitivity 19%–87%, specificity 56%–86%) maternal urine (2 studies; sensitivity 5%–15%, specificity 97%–100%), and biomarker test batteries (3 studies; sensitivity 22%–50%, specificity 56%–97%). Tests of the total concentration of 4 fatty acid ethyl esters (in meconium: 2 studies; in placenta: 1 study) demonstrated high sensitivity (82%–100%);
however, specificity was variable (13%–98%).
LIMITATIONS: Risk of bias was high due to self-report reference standards and selective outcome reporting.
CONCLUSIONS: Current evidence is insufficient to support the use of objective measures of prenatal alcohol exposure in practice. Biomarkers in meconium and placenta tissue may be the most promising candidates for further large-scale population-based research.
... Downloaded from false-positive results to maximize early detection of asymptomatic conditions. 93 Early diagnosis and intervention are associated with improved outcomes for children with FASD, 43 and thus it could be argued that a test with high sensitivity could be favored over specificity in this context because it may facilitate appropriate monitoring and follow-up among children with positive PAE screens. However, PAE is a highly emotive topic, and false-positive errors may lead to stigmatization and unnecessary burden on health care resources, and may even be used in legal proceedings against mothers. ...
... Given the implications of both types of diagnostic error, various authors have suggested that both high sensitivity and specificity are a prerequisite for the introduction of PAE screening. 55,93,96 ...
... More research is required to establish which method is most feasible and acceptable to stakeholders, including clinicians, policy makers, and families. 93,94,107,108 Assay methods for the biomarkers included in this review were highly variable. This is likely to account for some of the observed heterogeneity in findings. ...
Aim Diagnosis of fetal alcohol spectrum disorders (FASD) is often complicated by missing or unreliable information about alcohol exposure in-utero. Objective measurement of prenatal alcohol exposure is essential for identifying children at risk of FASD to support diagnosis and early intervention. Biomarkers have been advocated for use in population based screening programmes and feature as a recommended method within the Canadian National Screening Toolkit for FASD. However, their validity has not been comprehensively evaluated. This study aimed to systematically review the validity of objective measures of prenatal alcohol exposure.
Methods We searched 13 electronic databases and supplementary sources to identify relevant studies published between January 1990 and October 2015. Eligible studies provided data about the validity of biomarkers within maternal and neonatal biological matrices for the detection of prenatal alcohol exposure. We excluded animal studies and non-English language publications. Methodological quality was assessed using the Quality Assessment of Diagnostic Accuracy Studies tool (QUADAS-2) and evidence was synthesised using narrative analysis.
Results Twelve studies met inclusion criteria. Most (8) studies investigated objective measures of heavy prenatal alcohol exposure and included pregnant women and neonates from high risk settings, such as substance misuse clinics. Test performance varied widely for biomarkers in meconium (7 studies, sensitivity 4% to 100%, specificity 13% to 98%), maternal blood (4 studies, sensitivity 0% to 100%, specificity 79% to 100%), maternal hair (2 studies, sensitivity 19% to 85%, specificity 79% to 86%) maternal urine (2 studies, sensitivity 15%, specificity 97% to 100%) and biomarker test batteries (2 studies sensitivity 22% to 50%, specificity 56% to 97%). Placental biomarkers demonstrated 82% sensitivity and 83% to 94% specificity in one study. Selected outcomes are presented in Figure 1.
Methodological quality assessment suggested a high risk of bias within included studies due to the use of self-report reference standards and selective outcome reporting (Figure 2).
Conclusions Current evidence is insufficient to support the use of objective measures for prenatal alcohol exposure screening in practice. Biomarkers in meconium and placental tissue may be the most promising candidates for future research and require validation in population based studies.
... Secondary outcomes of FASD include adverse BPSSC characteristics including psychosocial and behavioural disabilities, learning difficulties, and mental ill-health that manifest as challenges with schooling, substance misuse, unemployment, and engagement with the criminal justice system (Bagley, 2019;Zizzo et al., 2013). The secondary outcomes of FASD are often "invisible" due to the presentation of co-occurring symptoms and overlapping behaviours that lead to misdiagnosis and a failure to include FASD as a complete diagnosis that encompasses a range of BPSSC characteristics. ...
... Targeted screening programs have failed to reduce the negative impact of stereotyping on families who experience PAE. For instance, marginalised communities, living in rural and remote areas where there are increased psychosocial and cultural-spiritual risk factors associated with substance misuse or where low-socioeconomic-status groups live, continue to be unequally targeted, over-represented, and stigmatised (Basaraba et al., 2016;Zizzo et al., 2013). ...
In Australia, it has been well documented that the leading preventable cause of nongenetic neurodevelopmental disability (NDD) is Fetal Alcohol Spectrum Disorder (FASD). This review explores literature informing Australian social work in the context of FASD. It highlights the need for increased social work research to inform evidence-based practice (EBP) in FASD diagnosis and management using the biopsychosocial-spiritual-cultural (BPSSC) framework. Social workers are often first to identify children's emotional, behavioural, and learning difficulties that may be a characteristic of FASD. Nonetheless, there is limited knowledge and understanding about how social workers in Australia address FASD. We argue that research about FASD and social work practice can improve social workers’ understanding of the BPSSC characteristics and management of FASD across the life course and contribute to EBP development in these areas in Australian social work.
• IMPLICATIONS
• Social workers have an emerging role in the holistic assessment, diagnosis, and management of FASD in Australia.
• Social workers are well placed in being first to recognise and identify an individual’s emotional, behavioural, sociocultural, and learning difficulties that are characteristics of FASD.
• Social workers can contribute to ongoing care in a tailored FASD management plan that links the caregivers to appropriate local community resources, service provision, and disability supports.
... This kind of regulation of women can be seen to grant certain legal rights to foetuses, where the rights of the foetus are deemed superior to those of the mother, and the state's assumed authority to protect the foetus overrules the interests of the mother (Meurk et al., 2014). Criminalisation of maternal substance use threatens women's autonomy, and the status of women as equal citizens who are entitled to protection under the law may further jeopardise infant health and wellbeing, may lead women to avoid antenatal care, may unfairly target already-marginalised groups, and does not necessarily prevent substance abuse among women (Zizzo, Di Pietro, Green et al., 2013). ...
... Universal screening is most respectful of the principle of justice and minimises the risk of stigmatising and stereotyping those population groups viewed to be at risk (Zizzo et al., 2013). It is recognised that many women under-report their AOD use during pregnancy due to stigma and the fear of adverse consequences (Haug et al., 2014). ...
... The issue of consent is often raised in terms of drug screening; which in the specific case of pregnancy is complicated because the drug use affects both the mother and neonate. Additionally, the ethical principal of "respect for persons" mandates that the woman give consent for the procedure given the social and legal ramifications of the test (Zizzo et al., 2013). Women also fear stigmatization and the legal consequences of drug use in pregnancy. ...
... As we have discussed, risk-based screening is not recommended because of the potential for bias. The ethical principal of justice may require that universal screening be done (Zizzo et al., 2013). However, in public health, screening is usually ethically differentiated as "with" or "without" consent (Moreno and Minkoff, 1992). ...
North America is currently suffering from one of the worst epidemics of illicit drug use in recent history: the opioid crisis. Pregnant women are not immune to the ravages of substance misuse which affects themselves, their pregnancies, and the wider community. The prevalence of drug misuse in pregnancy is not well quantified due to the lack of good validated tests, cooperation between clinicians and scientists developing tests, and consensus as to who should be tested and how results should be used. A wide range of tissues can be tested for drug use, including maternal blood, urine, and hair; neonatal meconium, urine, and hair; and placenta and umbilical cord tissues. Testing methods range from simple spectrophotometry and clinical chemistry to sophisticated analytical HPLC or mass spectrometry techniques. The drive for ever greater accuracy and sensitivity must be balanced with the necessities of medical practice requiring minimally invasive sampling, rapid turnaround, and techniques that can be realistically utilized in a clinical laboratory. Better screening tests have great potential to improve neonatal and maternal medical outcomes by enhancing the speed and accuracy of diagnosis. They also have great promise for public health monitoring, policy development, and resource allocation. However, women can and have been arrested for positive drug screens with even preliminary results used to remove children from custody, before rigorous confirmatory testing is completed. Balancing the scientific, medical, public health, legal, and ethical aspects of screening tests for drugs in pregnancy is critical for helping to address this crisis at all levels.
... However, other genomic elements including repetitive elements, retro-transposons and imprinted genes (wherein one parent-of-origin allele is silenced while the allele derived from the other parental genome is transcribed), are expected to exhibit similar methylation patterns across tissue and their assessment may permit inferences about the effects of environmental agents on other tissues including the brain. Loss of imprinting in particular is associated with fetal growth retardation or overgrowth syndromes, and therefore assessment of the imprinted status of these gene loci is likely to be particularly informative about child development and health (Fleisch, Wright, & Baccarelli, 2012). ...
... Evidence indicates support for universal screening under the above principles, and in the Canadian health care system, projected savings per quality-adjusted life year would justify the additional costs of routine testing. Finally, the limitations to the practice of bio-ethical principles were discussed, including practical limitations of 'opt-in' vs. 'opt-out' screening protocols, the inadequacy of counseling and disclosure practices in the medical system, limited access to services and tension in the medicalelegal profession between punitive and non-punitive approaches subsequent to diagnosis (Zizzo et al., 2013). ...
The 2012 meeting of the Fetal Alcohol Spectrum Disorders Study Group (FASDSG) focused on the development and ethics of biomarkers for fetal alcohol exposure. This one-day international conference brought students and trainees together with clinicians and researchers to discuss the latest research on FASD. One keynote speaker discussed the value of profiling epigenetic modifications in readily available fetal tissues to diagnose fetal exposure to environmental agents, while the second speaker discussed the ethics of biomarker development within the context of core principles of justice, autonomy, beneficence and non-maleficence. Three sessions of short data talks informed the audience of research advances with particular emphasis on the diagnosis of FASD. Other activities included updates on FASD-related activities by representatives of government agencies, a report on the implementation FASD-related diagnostic criteria in the fifth edition of the Diagnostic and Statistical Manual (DSM-5) of the American Psychiatric Association and a networking lunch, and the presentation of the "Merit Award" to Dr. Nathan Muraski for his work on behavioral outcomes of fetal alcohol exposure. The capstone of the meeting was the presentation of the "Henri Rosett" award to Dr. Denis Viljoen, in recognition of his role in raising awareness about the incidence of FASD in South Africa and in promoting FASD prevention and treatment programs as chairperson and chief executive officer of the Foundation for Alcohol Related Research (FARR).
... The ethical principal of autonomy mandates that patients give consent for a urine drug screen given the potential social and legal ramifications of the screen (Zizzo et al., 2013). According to the Guttmacher Institute, 23 states and the District of Columbia currently consider substance use during pregnancy to be child abuse under child-welfare statutes, and 3 states consider it grounds for civil commitment (Guttmacher Institute, 2021). ...
Introduction
This study evaluates racial and ethnic differences in urine drug screening and patient consent to urine drug screening at a single tertiary care center.
Methods
We conducted a retrospective cohort study of all deliveries at a single tertiary care center from January 1, 2015 to December 31, 2019. Medical records were queried for demographic data, performance of urine drug screening, commonly used diagnoses that prompted screening, documentation of patient consent, and result of screen. Associations between these outcomes were then assessed using Chi-square analysis and logistic regression.
Results
During the study period, 685 of 9953 (6.9%) of patients had a urine drug screen performed. Non-Hispanic Black patients comprised 33.6% of patients receiving screening, but only 16.6% of the total population. Of examined indications for urine drug screening, only insufficient prenatal care and trauma differed significantly between groups. After adjusting for commonly used diagnoses prompting screening, non-Hispanic black patients were significantly more likely to have urine drug screening performed (OR 2.0, 95% CI 1.6–2.4). Non-Hispanic Black and Hispanic patients were not significantly more likely to have a positive screen result when compared to Non-Hispanic White patients. Consent to urine drug screening was poorly documented (only 11.7% of patients had documented consent). This did not differ significantly between the major racial or ethnic groups.
Conclusion
Non-Hispanic Black and Hispanic patients experience differences in urine drug screening during admission for delivery that cannot be solely explained by differences in incidence of diagnoses that typically trigger screening. Documentation of patient consent to urine drug screening is poor.
... Two columns formed the diary: one for the alimentary behaviors during the working days and the other column for the weekend. In general, it is recommended to include questions about drinking in the context of a diet diary to avoid direct requests about alcohol consumption [68]. Pregnant women were asked about their eating habits during breakfast, lunch, and dinner as follows: ...
Background
Ethyl glucuronide (EtG) is a metabolite of ethanol used as a marker of alcohol drinking and is identified in urine. Gestational alcohol drinking harms the fetus, so, disclosing any form of use and abuse of this substance during pregnancy is crucial. Many discovery methods have been planned to overcome this question, including that of using screening questionnaires as the AUDIT-C, T-ACE/TACER-3, and TWEAK.
Aim
The aim and novelties of this study were to compare biochemical data from urinary EtG assays (cut-off 100 ng/mL for risking drinking behavior) with the outcome of questionnaires and of a food diary routinely used in our hospital; moreover, for the first time, we analyzed in pregnant women the EtG values normalized by the amount of creatinine excreted according to methods previously established [1].
Methods
Random urine samples were collected from 309 pregnant women immediately after being interviewed. EtG was quantified using an enzyme immunoassay and urinary creatinine was assessed using an enzymatic colorimetric method. Women that had not exhaustively answered one of the questionnaires, or that refused to provide urine samples were excluded. In the end, 309 women had a complete set of data and were considered for this study. Urine creatinine measurements were performed to determine if urine dilution might have resulted in false negatives in the challenge study. In order to accomplish this objective, as urinary creatinine concentrations are, on average, approximately 1 mg/mL, we used a normalized value of 100 ng EtG/mg Creatinine [1].
Results
Our data show that 20.4% of the pregnant women in the study were over the established normalized cut-off value. Poor to null concordance (unweighted k < 0.2) was found between EtG data and the screening interviews, that show, on average, lower levels of alcohol consumption.
Conclusion
In conclusion, this study provides evidence that the assessment of maternal alcohol consumption during pregnancy, only indirectly estimated with questionnaires and food diary, can produce misleading ratings.
... TA B L E 4 Quality assessment of studies using the Quadas-2 framework logical parents, and women in particular, whose children are identified as having FASD also require consideration (Maxim et al., 2014;Miranda et al., 2013;Zizzo et al., 2013). Additionally, the follow-up assessment and care needs required following a positive FASD screen can be expensive and time-consuming, potentially contributing to misuse of limited resources (Clarren et al., 2011;Maxim et al., 2014;Popova et al., 2013Popova et al., , 2020. ...
Background
Fetal alcohol spectrum disorder (FASD) is a prevalent neurodevelopmental disorder that is caused by prenatal alcohol exposure (PAE) and associated with a range of cognitive, affective, and health concerns. Although the identification of FASD can facilitate the provision of interventions and support, and plays a protective role against adverse outcomes, there are high rates of missed detection. The identification of FASD via screening may improve its recognition across settings. The current systematic review examined the available evidence on FASD screening tools and approaches across age groups and settings.
Methods
A systematic search was carried out for both peer‐reviewed studies and gray literature sources published between January 1990 and May 2020 and was preregistered with PROSPERO (#CRD42019122077). Studies included in the review focused on human applications of FASD screening in children, adolescents, and adults. The quality of the studies was assessed using the QUADAS‐2 and GRADE frameworks.
Results
The search yielded 20 screening tools and approaches across 45 studies, broadly characterized in 2 groups. The first group included approaches currently in use that aim to identify individuals at risk of FASD using a range of markers (n = 19) or associated sentinel dysmorphic facial features (n = 6). Another group of studies, characterized as emerging, focused on identifying promising biomarkers of PAE/FASD (n = 20). Overall, we identified limited research supporting the psychometric properties of most screening approaches. The quality review provided evidence of bias due to the common use of case–control designs and lack of adequate reference standards.
Conclusions
Although several FASD screening tools and approaches are available for use across a range of age groups and settings, the overall evidence base supporting their psychometric properties is weak, with most studies demonstrating significant risk of bias. Service providers should exercise caution in selecting and implementing FASD screening tools given these limitations. It is critically important to accurately identify individuals with FASD across ages and settings to support healthy outcomes. Thus, there is a pressing need for additional research in this area, particularly validation studies in large and representative samples using robust methodological approaches.
... Furthermore, the social ramifications of an unexpected positive are great and include increased risk of maternal stigmatization, criminalization, and mother-child separation. (4,5,8,9) Indications for toxicology testing are subjective, and no prior studies exploring evidence-based recommendations exist. ...
Background
Despite illicit substance use in pregnancy occurring across all demographic groups, minority and lower income pregnant and delivering patients tend to undergo testing at a higher rate than their counterparts. National guidelines for indications do not exist, and ordering of toxicology testing may be applied inequitably.
Objective
To evaluate whether any documented indications in a large cohort of patients were associated with a positive toxicology test, and whether indications for urine toxicology testing were applied consistently to different demographic groups.
Study Design
Retrospective cohort study reviewing pregnant and delivering patients who underwent toxicology testing on obstetric units at one institution from 5/30/2015 to 12/31/2018. Age, race, marital status, median income of residential zip code, indications for testing, and test result were collected for each patient by individual chart review. Indications included preterm complications (preterm prelabor rupture of membranes or preterm labor), abruption or hypertension, reported substance use, fetal complications, maternal complications, and none. Multivariate logistic regression models were analyzed for association between indication and test result, and for likelihood of marijuana as the sole positive test result. Logistic regression was used to evaluate the relationship of indication for testing with maternal race.
Results
Among 20,274 births, 551 patients underwent toxicology testing during the study period. No indication for drug toxicology testing was associated with a positive result except reported current or prior substance use. Compared to White patients, Black and Hispanic women were 4.26 (95%CI: 2.55, 7.09) and 5.75 (95%CI: 2.89, 11.43) times more likely to have toxicology testing for an indication other than reported substance use. Of all patients with positive tests (n=194), 48% tested positive for marijuana only.
Conclusions
Compared to their White counterparts, Black and Hispanic pregnant and delivering patients may be more frequently toxicology tested for indications less clearly associated with illicit substance use. Absence of evidence- based guidelines for toxicology testing on obstetrics units risks inequitable care and stigmatization of patient groups.
... A growing body of literature, including our findings, advocates for the combined use of self-reported measures and ethanol biomarkers (Bakhireva and Savage, 2011); however, self-report remains the most widely used and accessible method to screen for alcohol use during routine prenatal care. Acknowledging the ethical and legal issues associated with screening for substance use in pregnancy (Zizzo et al., 2013), which are beyond the scope of this report, the aim of the current study was to compare the prevalence of self-reported alcohol use obtained using different approaches. ...
Background
Accurate characterization of prenatal alcohol exposure (PAE) is challenging due to inconsistent use of screening questionnaires in routine prenatal care and substantial underreporting due to stigma associated with alcohol use in pregnancy. The aim of this study was to identify self‐report tools that are efficient in accurately characterizing PAE.
Methods
Participants meeting eligibility criteria for mild‐to‐moderate PAE were recruited into the University of New Mexico Ethanol, Neurodevelopment, Infant and Child Health cohort (N = 121) and followed prospectively. Timeline follow‐back (TLFB) interviews were administered at baseline to capture alcohol use in the periconceptional period and 30 days before enrollment; reported quantity was converted to oz absolute alcohol (AA), multiplied by frequency of use and averaged across 2 TLBF calendars. The interview also included questions about timing and number of drinks at the most recent drinking episode, maximum number of drinks in a 24‐hour period since the last menstrual period, and number of drinks on “special occasions” (irrespective of whether these occurred within the TLFB reported period). Continuous measures of alcohol use were analyzed to yield the number of binge episodes by participants who consumed ≥4 drinks/occasion. The proportion of women with ≥1 binge episode was also tabulated for each type of assessment.
Results
Average alcohol consumption was 0.6 ± 1.3 oz of AA/day (≈ 8.4 drinks/wk). Only 3.3% of participants reported ≥1 binge episode on the TLFB, 19.8% had ≥1 binge episode when asked about “special occasions,” and 52.1% when asked about the number of drinks the last time they drank alcohol. An even higher prevalence (89.3%) of bingeing was obtained based on the maximum number of drinks consumed in a 24‐hour period.
Conclusions
Self‐reported quantity of alcohol use varies greatly based on type of questions asked. Brief targeted questions about maximum number of drinks in 24 hours and total number of drinks during the most recent drinking episode provide much higher estimates of alcohol use and thus might be less affected by self‐reporting bias.
... Such fears can discourage them from seeking prenatal care altogether. 12,18,19 An additional issue in considering screening instruments for substance use disorders is that the validity, reliability, and clinical utility of standardized questionnaires that are used in screening for illicit drug use have received only limited evaluation in pregnancy. 20 Many tools that are used outside of pregnancy attempt to identify individuals with a substance use disorder. ...
... In general, it is recommended to include questions about drinking in the context of a diet diary, always for the purpose of avoiding social stigma. 49 Results of the interview are available in Table 3. ...
Fetal alcohol spectrum disorders (FASDs) are a group of negative conditions occurring in children exposed to alcohol during gestation. The early discovery of FASD is crucial for mother and infant follow‐ups. In this study, we investigated in pregnant women the association between urine ethylglucuronide (EtG—a biomarker of alcohol drinking) and indicators of the physical characteristics of FASD by prenatal ultrasound in the second trimester of gestation. We also correlated these data with the AUDIT‐C, T‐ACE/TACER‐3, TWEAK, and food habit diary, screening questionnaires used to disclose alcohol drinking during pregnancy. Forty‐four pregnant women were randomly enrolled and examined for ultrasound investigation during the second trimester of gestation. Urine samples were provided by pregnant women immediately after the routine interviews. EtG determinations were performed with a cutoff established at 100 ng/mL, a value indicating occasional alcohol drinking. Fifteen of the enrolled pregnant women overcame the EtG cutoff (34.09%). Analysis of variance (ANOVA) revealed that the fetuses of the positive EtG pregnant women had significantly longer interorbital distance and also significantly increased frontothalamic distance (P's < 0.02). Quite interestingly, no direct correlation was found between EtG data and both food diary and AUDIT‐C. However, a significant correlation was observed between urinary EtG and T‐ACE (r = 0.375; P = 0.012) and between urinary EtG and TWEAK (r = 0.512; P < 0.001) and a concordance with all questionnaire for EtG values higher than 500 ng/mL. This study provides clinical evidence that the diagnosis of maternal alcohol consumption during pregnancy by urine EtG may disclose FASD‐related damage in the fetus.
... • there are few FASD screening policies for newborns (Zizzo et al., 2013); • individuals are commonly diagnosed later in life when symptoms such as learning disabilities and challenging behaviors become apparent (Hellemans, Sliwowska, Verma, & Weinberg, 2010); • diagnostic assessments include disclosure from birth mothers that alcohol was consumed during their pregnancy (Chudley et al., 2005); • FASD diagnostic teams are not available in many parts of Canada (Clarren & Lutke, 2008); • how an FASD diagnosis will improve access to needed supports and services are highly varied; and • most people who have been diagnosed are children in care, adults in the correctional system, and Indigenous communities that have self-identified FASD as an area of concern (Badry, 2009(Badry, , 2010MacPherson & Chudley, 2007;Public Health Agency of Canada, 2012;Salmon, 2010). ...
Objectives
In this paper, we provide an overview of best practices in FASD prevention, diagnostic, and interventions and supports. In Canada, people diagnosed with Fetal Alcohol Spectrum Disorder (FASD) represent a fraction people living with FASD. While social stigma may deter people from seeking an FASD diagnosis, other deterrents include the lack of screening and diagnostic referrals, cost of travelling to a clinic, and lack of clarity of how a diagnosis may improve supports and services. Preventing FASD and improving lifelong outcomes for people living with FASD requires a coordinated approach between prevention, diagnostic, intervention, and support efforts.
Methods
Using the example of Newfoundland and Labrador, a province where 60% of the population lives in rural communities and benefits from being involved in national initiatives and partnerships, we discuss efforts underway in other Canadian provinces to address FASD.
Results
We make three recommendations that begin to address FASD‐specific needs in both rural and urban regions: a) a provincial FASD consultant position, b) an explicit partnership between provincial government and fasdNL, and c) increased access to FASD diagnostic teams.
Conclusion
While the recommendations are both modest and essential first steps, we also suggest that collaborations and resource‐sharing in FASD prevention and supports are more about doing things differently, rather than doing more.
... 73,79 Target population groups may avoid disclosing accurate information or participating in research or measuring maternal alcohol consumption through biomarker testing or interviews and surveys due to concerns of negative stereotyping, 80 fear of reprisals, 81 and a perceived lack of benefit of the detection of alcohol consumption and FAS diagnosis. 82 An assessment of the willingness of women at risk to participate in a meconium fatty acid ethyl ester (FAEE) screening program found low participation rates, and this was attributed to possible maternal, ' . . . embarrassment, guilt, fears of stigmatization and child apprehension (despite assurance otherwise).' 81 ...
Objective:
Fetal Alcohol Spectrum Disorder (FASD) is a preventable disorder caused by maternal alcohol consumption and marked by a range of physical and mental disabilities. Although recognized by the scientific and medical community as a clinical disorder, no internationally standardized diagnostic tool yet exists for FASD.
Methods and results:
This review seeks to analyse the discrepancies in existing diagnostic tools for FASD, and the repercussions these differences have on research, public health, and government policy.
Conclusions:
Disagreement on the adoption of a standardised tool is reflective of existing gaps in research on the conditions and factors that influence fetal vulnerability to damage from exposure. This discordance has led to variability in research findings, inconsistencies in government messaging, and misdiagnoses or missed diagnoses. The objective measurement of the timing and level of prenatal alcohol exposure is key to bridging these gaps; however, there is conflicting or limited evidence to support the use of existing measures.
... Of these, blood sampling may be the most promising, given that sampling blood is acceptable to most cultures, sample preparation is relatively straight forward and there are many alcohol metabolites and markers for alcohol consumption which remain within the blood for an extended period (Bakhireva and Savage, 2011). It is important to recognise that the use of biological samples for screening raises wider ethical issues related to consent, stigmatisation, individual rights and the rights of the unborn child (Mizejewski, 2010;Zizzo et al., 2013). ...
Providing appropriate antenatal and postnatal care for women who drink alcohol in pregnancy is only possible if those at risk can be identified. We aimed to compare the prevalence of alcohol consumption in the first trimester of pregnancy using self-report and blood biomarker analysis. Six-hundred routine blood samples from 2014, taken at the antenatal booking appointment, in the first trimester of pregnancy, were anonymously analysed for the presence of Carbohydrate Deficient Transferrin (CDT), a validated marker of chronic alcohol exposure (normalising 2-3 weeks from abstinence) and Gamma-glutamyltransferase (GGT), a liver enzyme elevated for up to 8 weeks after alcohol exposure. In a separate sample of women, from 2015, data taken during the antenatal visit, documenting women's self-reported alcohol consumption, were collected. The percentage of women who reported alcohol intake in the first trimester was 0.8%. This compared to 74.1% of women who reported consuming alcohol before pregnancy. CDT analysis revealed a prevalence rate of 1.4% and GGT a prevalence rate of 3.5% in the first trimester of pregnancy. Although those with elevated CDT generally had high levels of GGT, only one person was positive for CDT and GGT. Results from CDT analysis and self-report may underestimate prevalence for different reasons. GGT appeared to lack specificity, but it may have value in supporting findings from CDT analysis. Further studies using additional blood biomarkers, or a combination of blood biomarkers and self-report, may be beneficial in accurately detecting alcohol drinking history in pregnancy.
... Being proactive in discussions of alcohol use with male and non-pregnant female patients of all backgrounds is also important to reducing the many potential harms of alcohol consumption, and this universal approach may reduce feelings of being targeted or stigmatized. 49 ...
p>Fetal alcohol spectrum disorder (FASD) is a leading form of neurodevelopmental delay in Canada, affecting an estimated 3000 babies per year. FASD involves a range of disabilities that entail significant costs to affected individuals, families, and society. Exposure to alcohol in utero is a necessary factor for FASD development, and this has led to FASD being described as “completely preventable”. However, there are significant ethical challenges associated with FASD prevention. These challenges revolve around 1) what should be communicated about the risks of alcohol consumption during pregnancy, given some ongoing scientific uncertainty about the effects of prenatal alcohol exposure, and 2) how to communicate these risks, given the potential for stigma against women who give birth to children with FASD as well as against children and adults with FASD. In this paper, we share initial thoughts on how primary care physicians can tackle this complex challenge. First, we recommend honest disclosure of scientific evidence to women and the tailoring of information offered to pregnant women. Second, we propose a contextualized, patient-centred, compassionate approach to ensure that appropriate advice is given to patients in a supportive, non-stigmatizing way.</p
... Ethical principles and guidelines for the protection of human subjects of research," 2014), i.e., Respect for persons, Beneficence, and Justice, can serve as a basis for such regulations. As comprehensively reviewed by the bioethicists and FASD researchers in Canada (Zizzo et al., 2013), important considerations in PAE screening are the consent to screening and confidentiality (Respect), cost-effectiveness, appropriate follow-up, and availability of the treatment services (Beneficence, Justice), and decriminalization and de-marginalization of individuals with PAE, as well as equitable access to available resources (Justice). While early diagnosis and services for children with FASD in the welfare system are essential for reducing placement disruption (Alex & Feldmann, 2012;Olson, Oti, Gelo, & Beck, 2009), our study highlights an astonishing paucity of resources for families living in underserved areas, as PDC is the only resource offering FASD evaluations for approximately 70 surrounding counties. ...
Fetal Alcohol Spectrum Disorders (FASD) might be 10–15 times more prevalent among foster/adopted children compared to the general population; however, many of these children remain undiagnosed or misdiagnosed. The lack of confirmed prenatal alcohol exposure (PAE) may be a key barrier to diagnosis. Our sample included 681 patients evaluated for FASD, according to the University of Washington 4-Digit Diagnostic Code, at a pediatric specialty clinic. Guardianship status and other patient characteristics were evaluated by multinomial logistic regression as potential predictors of being classified into one of the following FASD groups: 1) full or partial Fetal Alcohol Syndrome (FAS/pFAS; n = 97); 2) Static Encephalopathy/Alcohol-Exposed (SE/AE) or Neurobehavioral Disorder/Alcohol-Exposed (ND/AE) (n = 135); and 3) some features of FASD (equivalent to pFAS, SE/AE or ND/AE phenotypes) but unknown PAE (n = 449). Median age at assessment was 7.0 years, non-Hispanic White constituted the predominant racial/ethnic group (49.5%), and the majority (81.8%) lacked involvement from a biological parent/relative. Many patients (66.0%) had some features of FASD but lacked reliable PAE information. Children classified into the ‘some features/unknown PAE’ group had higher median age of assessment (8 years) compared to other groups (6 years; p < 0.001). No association was observed between race/ethnicity or child’s sex and FASD outcomes (p > 0.05). Adopted/foster children were 2.8 times as likely (95% CI: 1.6; 4.8) to be classified into the ‘some features/unknown PAE’ group compared to children living with a parent/relative after adjusting for covariates. This study’s findings indicate that adopted/foster children are more likely to have unknown PAE and not receive a FASD diagnosis, potentially denying them access to specialized services, treatment, and rehabilitation.
... If the women assumes alcohol (positive score), the investigators ask the specification of drink size for different types of alcoholic beverages. In general, it is recommended to imbibe questions about drinking in the context of a diet diary, always on the purpose of avoiding social stigma (Zizzo et al., 2013). Results of the interview are available in Table 3. ...
Ethyl glucuronide (EtG) is an ethanol metabolite and EtG is used as a biomarker of alcohol drinking. EtG can be detected in the blood and in several biological matrices including urine, hair and nails. Alcohol consumption during pregnancy is a strong risk factor for fetus health so in the recent years different strategies to reveal alcohol use have been planning including the use of screening questionnaires as the AUDIT-C, T-ACE and TWEAK. The present study aims to investigate in pregnant women the specificity and predictive value of the AUDIT-C, T-ACE and TWEAK plus a food diary in use in Sapienza University Hospital compared with the results of urine EtG measurement. Seventy pregnant women were enrolled and examined. Urine samples were provided by pregnant women immediately after the interviews. EtG determinations were performed by Enzyme Immunoassay with a cut-off established at 100 ng/mL.
... Any type of screening for highly stigmatized conditions is fraught with ethical challenges (Zizzo et al., 2013). Factors such as fear of prosecution, child care needs, financial stress, and social stigma may present significant barriers for even the most motivated woman struggling with alcohol dependence to seek treatment during or after pregnancy. ...
Background:
While 2 to 5% of school-aged children in the United States are estimated to be affected by fetal alcohol spectrum disorders (FASD), the prevalence of prenatal alcohol exposure (PAE) might be substantially underreported. Our objective was to systematically estimate the prevalence of PAE in Texas by measuring a direct ethanol metabolite, phosphatidylethanol (PEth), in 1,000 infant residual dried blood spots (irDBSs) in the Texas Newborn Screening Repository.
Methods:
All public health regions (PHRs) were represented proportional to their 2014 birth rate (~0.25% of total births). A cross-sectional study design (unit of observation: individual irDBS cards/infants) with additional ecologic subanalysis (unit of observation: aggregate measures for each Texas PHR) was utilized. The study used PEth-irDBS to estimate the prevalence of PAE within 1 month before delivery for the state of Texas and each Texas PHR. The ecologic subanalysis compared different geographical regions' aggregate prevalence of PAE with (i) retail liquor licenses, (ii) median household income by PHR, and (iii) prevalence of birth outcomes commonly associated with FASD.
Results:
The sample included an equal number of males and females; 47.8% non-Hispanic White, 40.8% Hispanic, 6.6% African American, and 4.8% Asian infants. In the entire sample, 8.4% of irDBSs were positive for PEth (>20 ng/ml) indicative of PAE within approximately 1 month before delivery. Large regional differences were observed with mostly urban, high median-income regions demonstrating the highest prevalence.
Conclusions:
Results of this first systematic statewide PAE prevalence study demonstrate that PAE might be more prevalent than previously thought. Active case ascertainment efforts for FASD coupled with systematic objective assessment of PAE should expand to the national level to better estimate public health needs required to provide adequate services for children affected by PAE.
... Pediatricians or mental health professionals may not be adequately trained on how to integrate information regarding prenatal alcohol exposure into their practice or the information regarding prenatal exposure may not readily available (Gahagan et al. 2006;Rojmahamongkol et al. 2015). Further, the diagnosis may be stigmatizing and thus providers may be hesitant to discuss it with the family (Zizzo et al. 2013). ...
This grand rounds manuscript reviews important considerations in developing case conceptualizations for individuals with a history of prenatal alcohol exposure. This case study provides an introduction to fetal alcohol spectrum disorders, diagnostic issues, a detailed description of the individual’s history, presenting symptoms, neuropsychological test results, and an integrated summary. We describe a 9-year-old girl diagnosed with a fetal alcohol spectrum disorder (FASD): Neurobehavioral Disorder Associated with Prenatal Alcohol Exposure (ND-PAE). This patient is a composite of a prototypical child who participated as part of a research project at the Center for Behavioral Teratology who was subsequently seen at an outpatient child psychiatry facility.
... Finally, there are a number of ethical, legal, and social implications when screening for PAE, as identification of PAE can result in the removal of the child from their mother and also her criminal prosecution (Zizzo et al., 2013). Given the extent of public misinformation about the safety of PAE and how it results in FASD (Singh et al., 2014), any screening for PAE should be used to only provide the appropriate therapies and educational aids for the child, which are most effective the earlier they are received, and not be used to persecute the past. ...
Rodent models of Fetal Alcohol Spectrum Disorders (FASD) have revealed that prenatal alcohol exposure (PAE) results in differential DNA cytosine methylation in the developing brain. The resulting genome-wide methylation changes are enriched in genes with neurodevelopmental functions. The profile of differential methylation is dynamic and present in some form for life. The methylation changes are transmitted across subsequent mitotic divisions, where they are maintained and further modified over time. More recent follow up has identified a profile of the differential methylation in the buccal swabs of young children born with FASD. While distinct from the profile observed in brain tissue from rodent models, there are similarities. These include changes in genes belonging to a number of neurodevelopmental and behavioral pathways. Specifically, there is increased methylation at the clustered protocadherin genes and deregulation of genomically imprinted genes, even though no single gene is affected in all patients studied to date. These novel results suggest further development of a methylation based strategy could enable early and accurate diagnostics and therapeutics, which have remained a challenge in FASD research. There are two aspects of this challenge that must be addressed in the immediate future: First, the long-term differential methylomics observed in rodent models must be functionally confirmed. Second, the similarities in differential methylation must be further established in humans at a methylomic level and overcome a number of technical limitations. While a cure for FASD is challenging, there is an opportunity for the development of early diagnostics and attenuations towards a higher quality of life.
... In any case, most relevant ethical aspects should be kept in mind [29] and need to be discussed before the implementation of screening programs. Examples given are consent and respect of persons, stigma and participation rates, the cost-benefit analysis of a screening program, consequences of false-positive and false-negative test results, confidentiality and appropriate follow-up to positive screening results, as well as the use of screen results for criminal prosecution [30]. Also the right to not know [31] should be considered. ...
... The ethical and legal issues raised by diagnosing FASD on the basis of self-reported maternal drinking during pregnancy also apply to the use of putative biomarkers, such as meconium tests, for detecting prenatal exposure to alcohol in the fetus or newborn baby [55,56]. These tests may establish if a child has been exposed to alcohol in utero more accurately than women's selfreported drinking but they raise similar considerations over the comparative 'rights' of the mother and her child. ...
The diagnosis of Fetal Alcohol Spectrum Disorders (FASDs) is embedded in a matrix of biological, social and ethical processes, making it an important topic for crossdisciplinary social and ethical research. This article reviews different branches of research relevant to understanding how FASD is identified and defined and outlines a framework for future social and ethical research in this area. We outline the character of scientific research into FASD, epidemiological discrepancies between reported patterns of maternal alcohol consumption during pregnancy and the incidence of FASD, and the social and ethical considerations that may impact on who is, and is not, diagnosed. We highlight what further research investigating FASD diagnostic processes, as well as the multi-generational impacts of FASD, is needed. Important research priorities are to: 1) enumerate the variety of stakeholders involved in seeking FASD diagnoses; 2) understand the experiences and perspectives of mothers from different backgrounds who have consumed alcohol during pregnancy and their affected children; and 3) collect health histories of maternal alcohol consumption in families to determine the effect of FASD at sub-cultural and cultural levels.
... Several issues remain to be investigated before PEth-DBS can be recommended as a reliable method for PAE assessment in the newborn period. Several recent studies (Mizejewski, 2010, Zizzo et al., 2013, including ours (Bakhireva et al., 2013), have brought up ethical issues of newborn testing for PAE, acceptability by patients, and conditions for ethical implementation of such testing, including maternal consent. Second, the stability of PEth-DBS is largely unknown. ...
Background:
Accurate identification of prenatal alcohol exposure (PAE) in the newborn period offers an opportunity for early identification of children at risk of future neurocognitive problems and the implementation of interventional approaches earlier in life. PAE newborn screening by measuring phosphatidylethanol in dried blood spot (PEth-DBS) cards is feasible, logistically easier, and more cost-efficient compared with other biomarkers. However, the sensitivity and specificity of this method have yet to be established.
Methods:
This prospective cohort study examined validity of PEth-DBS among 28 infants with PAE and 32 controls relative to maternal self-report and other biomarkers. Pregnant women were recruited from a University of New Mexico clinic and followed to early postpartum period. The composite index, which was based on self-reported measures of alcohol use and allowed to classify subjects into PAE and control groups, was the criterion measure used to estimate sensitivity and specificity of PEth-DBS.
Results:
The study included large proportions of patients representing ethnic minorities (7.4% American Indian, 81.7% Hispanic/Latina), low education (54.2% <high school), and unplanned pregnancy (90.0%). No differences in sociodemographic characteristics, smoking or illicit drug use were observed among the study groups. The sensitivity of maternal biomarkers (gamma glutamyltranspeptidase [GGT], % carbohydrate-deficient transferrin [%CDT], urine ethyl glucuronide [UEtG], urine ethyl sulfate [UEtS]) was low (<15%) reflecting a moderate chronic or intermittent binge pattern of drinking in this cohort. PEth-DBS demonstrated 100% specificity and the highest sensitivity (32.1%) compared with other biomarkers. A battery consisting of maternal direct ethanol metabolites (UEtG, UEtS, PEth) and newborn PEth-DBS increased sensitivity to 50% without a substantial drop in specificity (93.8%).
Conclusions:
Newborn PEth-DBS is a highly specific biomarker and can facilitate accurate detection of PAE in conjunction with other biomarkers. Minimal invasiveness, ease of storage and transportation of DBS cards, absence of postcollection synthesis, cost savings, and potential integration with routine newborn screening are all unique advantages of this method.
Background
Screening for fetal alcohol spectrum disorder (FASD) has been identified as a promising approach to improve recognition, understanding and effective response to the unique needs of those with FASD in criminal legal settings. However, to date, there has been limited synthesis of relevant screening tools, indicators, or implementation considerations in this context.
Aims
The present review aimed to synthesise evidence and develop a conceptual framework for understanding how, when, why, for whom and by whom FASD screening tools, items and/or indicators and characteristics serve to accurately identify people with FASD in criminal legal contexts, with consideration of individual and system needs relevant to effective implementation and response.
Methods
A preregistered search was conducted using a modified realist review framework for both peer‐reviewed articles and grey literature. Included sources were available in English, which focused on individuals with prenatal alcohol exposure and/or FASD with criminal legal involvement and offered new empirical evidence. Sources were reviewed using the Quality Control Tool for Screening Titles and Abstracts by Second Reviewer framework, extracted using a structured coding form and narratively synthesised.
Results
The search yielded 52 sources, 11 FASD screening tools designed for or applied in criminal legal settings and 38 potential FASD indicators or characteristics relevant to identifying people who may have FASD in criminal legal settings, organised into six conceptually related domains. There was limited evidence supporting the psychometric properties of screening tools across populations or settings, though growing evidence highlights the promise of some instruments. Although few studies characterised potential considerations to be made when implementing a screening tool or approach, both system and individual level needs related to recognising and effectively responding to FASD in criminal legal contexts were identified, and findings revealed strong support among legal and clinical professionals regarding the need for FASD screening in these settings.
Conclusions
Findings of this review can be used to inform the development, selection, implementation and evaluation of FASD screening tools in criminal legal settings and underscore a continued need for enhanced resources, policy and cross‐sectoral response to better support the needs of people with FASD in the criminal legal contexts.
Prenatal exposure to alcohol can lead to harmful developmental outcomes and is the leading known cause of developmental disability in the western world. Fetal Alcohol Spectrum Disorder (FASD) describes the constellation of adverse effects that can result from maternal consumption of alcohol during pregnancy. The cognitive and behavioural symptoms associated with FASD are both debilitating and life-long. The extent and severity of these deficits require a high level of the coordinated management plan that involves service delivery and resource allocations that can be extremely costly to caregivers and the health care system. A timely diagnosis of FASD is the most important factor in the prevention of secondary disabilities, despite the inherent challenges of obtaining one. Unique approaches are needed to mitigate the impact of FASD and prenatal alcohol exposure, thus improving diagnosis, intervention and treatment for affected individuals.
An expanding policy framework aimed at monitoring alcohol consumption during pregnancy has emerged. The primary justification is prevention of harm from what is termed ‘prenatal alcohol exposure’ (PAE), by enabling more extensive diagnosis of the disability labelled fetal alcohol spectrum disorder (FASD). Here we focus on proposals to include biomarkers as a PAE ‘screening tool’, specifically those found in meconium (the first newborn excrement), which are discussed as an ‘objective’ measure of PAE.
We ask the overarching question, ‘Can routine screening of meconium to establish PAE be ethically or legally justified’, and we answer, ‘No’. To reach this conclusion, we discuss three areas. First, we consider the reasons why meconium screening should not be deemed ‘typical’ within the scope of accepted screening tools. We argue that given the aim and necessary timing of the screen, it cannot achieve what it promises. Second, we outline why patient autonomy and consent are not properly accounted for and cannot be reconciled with the ‘routinisation’ of the proposed ‘screening’. Last, we outline why the benefit of such a screen is not clear, focusing on the significance of trust in healthcare professionals (HCP) for the best interests of the future child and pregnant woman.
While recognising the adverse effects of heavy alcohol consumption during pregnancy, we emphasise the case for robust ethical, legal and social considerations and the central need for trust between HCP and patients in maternity care. We conclude the permissibility of meconium screening has not been proven, and it is not justified.
Articles published in Biomed Rev 2021; 32
Purpose
The purpose of this paper is to conduct a scoping review of the literature to explore the many ways stigma affects people with FASD and to highlight the disciplines and places where discourse on FASD and stigma is taking place.
Design/methodology/approach
Searches were conducted in PubMed, ERIC, Family & Society Studies Worldwide, Families Studies Abstracts and Google Scholar between 2008 and 2018. Search terms focused on stigma, shame and the connection to FASD with a view to looking across social and medical science literature.
Findings
Searches identified 39 full text manuscripts, 13 of which were included in the scoping review. Stigma toward people with FASD exists in multiple professional forums across disciplines. The relationship between mother’s use of alcohol and the lasting impact on the child is a focus in the articles identified from a public health perspective. The review showed there was limited cross-disciplinary discussion evident. In total 13 articles were selected for inclusion in this review.
Research limitations/implications
Negative discourses predominate with little attention being paid to possible areas of success as well as cases of lower FASD impacts. There is a significant void in work focusing on positive outcomes for people with FASD. Such discourse would support a better understanding of pathways to more positive outcomes.
Originality/value
This paper highlights the issue of FASD and stigma through identification of relevant literature and expands the conversation to offer insights into the challenging terrain that individuals with FASD must navigate. The issue of stigma is not linked only to individuals with FASD but also their support systems. It is critical to recognize the multiple attributions of stigma to FASD in order to effectively take up conversations across and between disciplines to promote new discourses focused on de-stigmatization.
Prenatal reporting to child protection services has been enacted into most jurisdictions across Australia and in other countries, its aims being to intervene early and provide supports which will either identify or prevent the need for a baby to be taken into care and protection once born. Despite indications that there are increasing numbers of prenatal reports, little is known about the characteristics of those reported, the timing and reasons for reports, service responses, and the impacts of being reported. This study is one of the first to use administrative data to examine the characteristics of two samples from one Australian jurisdiction: (i) data from casefiles of 38 cases reported in 2012-13, and (ii) administrative data from 117 cases reported prenatally in 2013. These data showed that women who were reported to child protection services in relation to their pregnancy were predominantly disadvantaged, and were likely to be reported relatively late in their pregnancy due to 'future risk concerns'. Approximately two-thirds of those reported were provided with some prenatal support, as recorded by the child protection system, generally of limited duration. Twelve percent of the babies born to the larger cohort of women were removed within 100days of their birth. It is likely that longer term supportive interventions are needed, to reduce the risk factors evident in women reported during pregnancy, and to improve their ability to safely care for their children. Information on the short and long-term impacts from rigorous evaluations and longer-term intervention trials are also vital to ensure that prenatal reporting and interventions are, in fact, improving outcomes for infants and families.
The fetal alcohol spectrum of disorders (FASD) includes four diagnostic categories for the clinical consequences of prenatal alcohol exposure (PAE) in the unborn child. Physical features are necessary for the diagnosis of the fetal alcohol syndrome (FAS) and partial pFAS. Moreover, these features are specific and a diagnosis of FAS can be made even in the absence of knowledge of PAE. Not only growth deficits, microcephaly and the 3 facial features (short palpebral fissures, smooth philtrum and narrow vermillion of the upper lip) are characteristic, since other dysmorphic features particularly in the hands are key to the recognition of FAS. Most features can be explained by the damage to the brain during pregnancy and can be replicated in animal models. Many different diagnostic guidelines are used for the diagnosis of FASD and the physical features are considered differently in each of them. There is a need for universal clinical criteria for the diagnosis of FASD if our goal is to favor universal recognition.
Background:
Neuroethics describes several interdisciplinary topics exploring the application and implications of engaging neuroscience in societal contexts. To explore this topic, we present Part 3 of a four-part bibliography of neuroethics' literature focusing on the "ethics of neuroscience."
Methods:
To complete a systematic survey of the neuroethics literature, 19 databases and 4 individual open-access journals were employed. Searches were conducted using the indexing language of the U.S. National Library of Medicine (NLM). A Python code was used to eliminate duplications in the final bibliography.
Results:
This bibliography consists of 1137 papers, 56 books, and 134 book chapters published from 2002 through 2014, covering ethical issues in neuroimaging, neurogenetics, neurobiomarkers, neuro-psychopharmacology, brain stimulation, neural stem cells, neural tissue transplants, pediatric-specific issues, dual-use, and general neuroscience research issues. These works contain explanations of recent research regarding neurotechnology, while exploring ethical issues in future discoveries and use.
Background
Fetal alcohol syndrome (FAS) is a well‐known consequence of prenatal alcohol exposure. However, women tend to deny or underreport their alcohol use during pregnancy. The aim of this study was to explore the usability of various alcohol biomarkers for FAS screening in a data set without information on self‐reported alcohol use.
Methods
A nationwide register study with a case–control design was conducted. The target population consisted of all live births in Finland from 1987 to 2005. FAS cases (n = 565) were identified from the Finnish Register of Congenital Malformations. Mothers of FAS cases and their controls were selected in a ratio of 1 to 2 from the Finnish Maternity Cohort (FMC). Background information was obtained from the Finnish Medical Birth Register. Serum samples, collected at the mother's first visit to the maternity care, were obtained from the national FMC biobank. Biomarkers of alcohol consumption, gamma‐glutamyltransferase (GGT), carbohydrate‐deficient transferrin (%CDT), combination of GGT and CDT (GGT‐CDT), and ethylglucuronide (EtG) were analyzed from mothers of FAS cases (n = 385) and their controls (n = 745).
Results
Median levels of all biomarkers were significantly higher among the mothers of FAS children than in control mothers. Using previously validated cutoffs for EtG, GGT, %CDT, and GGT‐CDT, nearly half (46%) of the mothers with affected offspring could be identified. The predictive association was highest for GGT‐CDT combination and significant also for all the other biomarkers.
Conclusions
In this explorative case–control study, we demonstrate that the FMC biobank can be used to screen alcohol biomarkers for epidemiological research purposes. According to our results, the use of alcohol biomarkers during the first trimester may help to identify the high‐risk pregnancies for FAS. A more systematic use of alcohol biomarkers at maternity care may open new possibilities for screening and intervention of alcohol use among pregnant mothers.
The mutual impact of Behçet's disease (BD) and pregnancy is variable and still unclear. Among the safe drugs administered, the newer infliximab (IFX) was rarely experienced in pregnancy, particularly in the third trimester.
The authors report a pregnancy with fetal growth restriction at 36 weeks in a 31-year-old primigravida with symptomatic BD, treated with uninterrupted monthly IFX and daily enoxaparin. The patient was induced at 38 weeks and had an uneventful vaginal delivery of a healthy baby. The postpartum period and following six months were uneventful for mother in terms of BD exacerbation, and newborn in terms of potential risks of neonatal BD and/or infections due to late immunosuppressive IFX administration.
Because of the inconstant mutual impact, BD pregnancies should be precautionary considered at "potential high-risk" and need a careful and close monitoring by a multidisciplinary team with specific expertise.
The use of alcohol and other substances is not infrequent during pregnancy and may be associated with adverse effects on pregnancy outcome. Many pregnant women may continue these practices throughout pregnancy and even after delivery, unless they are recognized and assessed. Screening may be one way to achieve consistent and early identification. Prenatal health care providers may wish to screen all pregnant patients for their use of alcohol and other drugs using an approach that works best in their setting. A positive screen is an opportunity for the clinician and patient to discuss health practices and behaviors.
A woman's alcohol use during pregnancy is one of the top preventable causes of birth defects and developmental disabilities that are known as fetal alcohol spectrum disorders (FASD). The social and economic burden of FASD is substantial. Lifetime direct tangible costs per individual related to health care, education and social services in Canada have been estimated to be $1.4 million. Screening women of child-bearing age and pregnant women and recording their alcohol consumption is a practical process to identify and evaluate women at-risk and to identify potentially exposed infants. The FASD Advisory Workgroup proposes the following three levels of screenings which should be done on consenting women: Level I screening involves practice-based approaches that can be used by health care providers when talking to women about alcohol use, such as motivational interviewing and supportive dialogue. Level II screening includes a number of structured questionnaires that can be used with direct questioning (TLFB) or indirect /masked screening (AUDIT, BMAST / SMAST, CAGE, CRAFFT, T-ACE, TWEAK). Level III screening includes laboratory-based tools that can be used to confirm the presence of a drug, its level of exposure and determine the presence of multiple drugs. There are challenges and limitations in the use of the screening and assessment tools outlined. For example, the single question about alcohol use and the various questionnaires rely on a woman to provide details about her alcohol use. There is no consensus on the appropriate screening to use across Canada as each provincial / territorial jurisdiction, health care organization and healthcare provider uses a variety of formal and informal screening tool. In addition, there are inconsistent processes across Canada for the recording of the alcohol use in a woman's chart and the transfer of the information to the infant and the child's health records. The FASD Advisory Workgroup proposes eleven recommendations to improve the screening and recording processes for alcohol use in women of child-bearing age and pregnant women.
The diagnosis of fetal alcohol spectrum disorder (FASD) is complex and guidelines are warranted. A subcommittee of the Public Health Agency of Canada's National Advisory Committee on Fetal Alcohol Spectrum Disorder reviewed, analysed and integrated current approaches to diagnosis to reach agreement on a standard in Canada. The purpose of this paper is to review and clarify the use of current diagnostic systems and make recommendations on their application for diagnosis of FASD-related disabilities in people of all ages. The guidelines are based on widespread consultation of expert practitioners and partners in the field. The guidelines have been organized into 7 categories: screening and referral; the physical examination and differential diagnosis; the neurobehavioural assessment; and treatment and follow-up; maternal alcohol history in pregnancy; diagnostic criteria for fetal alcohol syndrome (FAS), partial FAS and alcohol-related neurodevelopmental disorder; and harmonization of Institute of Medicine and 4-Digit Diagnostic Code approaches. The diagnosis requires a comprehensive history and physical and neurobehavioural assessments; a multidisciplinary approach is necessary. These are the first Canadian guidelines for the diagnosis of FAS and its related disabilities, developed by broad-based consultation among experts in diagnosis.
In this article, we compared the costs of testing meconium for alcohol exposure in newborns with the lifetime benefits of early detection and intervention.
A decision analytic model was developed to assess the cost-effectiveness of testing meconium for two scenarios: (1) all infants in the Canadian province of Ontario and (2) infants who have an older sibling diagnosed with fetal alcohol spectrum disorder (FASD). The model incorporated the costs of early screening, early intervention, and the lifetime societal benefits of early intervention.
The cost of the meconium test is Can. 1.3 million per incident case. The benefit of early intervention is an improvement in literacy, which improves the quality of life parameter by 0.17 and increases adult lifetime earnings by 65,874 per quality-adjusted life years. When considering targeted screening, there is a cost savings for society and improvements in quality of life.
Depending on society's willingness-to-pay threshold for improving infants' lives in a setting of considerable equity concerns, universal screening and targeted screening of infants who have an older sibling diagnosed with FASD both represent policies that are good value for the money.
National statistics indicate that 11% of newborns are exposed to drugs of abuse in utero (Chasnoff, Burns, Schnoll, & Burns, 1985). From July through November of 1993, 0.2% (1/583) of infants in our hospital screened positive for exposure to drugs of abuse in utero, by blood and urine screens collected per physician's order. During the same 4 months in 1995, 0.2% (1/574) screened positive. In contrast, from July through November 1994, all newborns born at our institution were screened for in utero drug exposure using meconium samples; approximately 4.5% (22/493) were positive. Meconium screening of all infants, not just those per physician order, produced a dramatic increase in positive drug results. Of great concern in this study was that only two of the 22 mothers identified as having drug-screen positive babies during the 1994 meconium screening agreed to use rehabilitative services. This highlights the necessity of intervention through early educational programs stressing prevention.
Meconium fatty acid ethyl esters (FAEEs) are validated biomarkers of fetal alcohol exposure. Meconium FAEE testing can potentially be used as a screen by health-care professionals to identify neonates at-risk for Fetal Alcohol Spectrum Disorder, thereby permitting diagnostic follow-up of these children and early intervention in those who develop disabilities. The purpose of this study was to assess whether women would willingly partake in a screening program of this nature. This was determined by launching a pilot screening program for prenatal alcohol exposure in a high-risk obstetric unit previously shown to have a high prevalence of FAEE-positive meconium via anonymous meconium testing. The program involved voluntary testing of meconium for FAEEs and long-term developmental follow-up of positive cases through an existing public health program. The participation rate in the screening program was significantly lower than when testing was conducted anonymously (78% vs. 95%, respectively; p < 0.05), and the positivity rate was 3% in contrast to 30% observed under anonymous conditions (p < 0.001). These low rates suggest that the majority of mothers who consumed alcohol in pregnancy refused to participate. We conclude that despite the potential benefits of such screening programs, maternal unwillingness to consent, likely due to fear, embarrassment, and guilt, may limit the effectiveness of meconium testing for population-based open screening, highlighting the need for public education and social marketing efforts for such programs to be of benefit.
The deleterious effects exerted by prenatal ethanol exposure include physical, mental, behavioural and/or learning disabilities that are included in the term fetal alcohol spectrum disorder (FASD). Objective assessment of exposure to ethanol at both prenatal and postnatal stages is essential for early prevention and intervention. Since pregnant women tend to underreport alcohol drinking by questionnaires, a number of biological markers have been proposed and evaluated for their capability to highlight gestational drinking behaviour. These biomarkers include classical biomarkers (albeit indirect) of alcohol-induced pathology (mean corpuscular volume (MCV), gamma glutamyltransferase (GGT), aspartate aminotransferase (AST) and alanine aminotransferase (ALT)) acetaldehyde-derived conjugates, and finally derivatives of non-oxidative ethanol metabolism (fatty acid ethyl esters (FAEEs), ethyl glucuronide (EtG), ethyl sulphate (EtS) and phosphaditylethanol (PEth)). Since ethanol itself and acetaldehyde are only measured few hours after ethanol intake in conventional matrices such as blood, urine and sweat, they are only useful to detect recent ethanol exposure. In the past few years, the non-oxidative ethanol metabolites have received increasing attention because of their specificity and in some case wide time-window of detection in non-conventional matrices from the pregnant mother (oral fluid and hair) and fetus-newborn (neonatal hair, meconium, placenta and umbilical cord). This article reviews bioanalytical procedures for the determination of these markers of ethanol consumption during pregnancy and related prenatal exposure. In addition, clinical toxicological applications of these procedures are presented and discussed.
Fatty acid ethyl esters (FAEEs) in meconium are validated biomarkers of heavy fetal alcohol exposure that may potentially be used clinically for identifying children at risk for alcohol-related disabilities. However, until now, FAEEs have been largely used anonymously in epidemiological studies, and by child protection authorities in need for verification of heavy alcohol use in pregnancy. Here we describe the first case of a neonate identified as part of a research study on a pilot neonatal screening program for prenatal alcohol exposure. The neonate's meconium tested high for FAEEs (52 nmol/g; positive cut-off ≥ 2 nmol/g), which prompted active follow-up of the infant's development, identifying early neurocognitive problems and allowing initiation of a remedial program.
Unlabelled:
In Canadian law, pregnant women are held to owe no enforceable duties of care to their children before birth, but healthcare providers may be held accountable once children are born alive for causing injuries prenatally. When children are born in hospitals, recovered meconium may be tested without consent, but there may be an ethical duty to inform mothers. Meconium belongs to the newborns, and mothers may be required to make decisions about its use in their children's best interests. Proposals to test meconium from particular populations raise concern about stigmatization.
Keywords:
Meconium, alcohol, fatty acid ethyl esters, ethics, legal duties, pregnancy, fetal alcohol spectrum disorder.
The objective of this report is to estimate the benefits of universal meconium screening for maternal drinking during pregnancy. Fetal alcohol spectrum disorder (FASD), including its most severe manifestation fetal alcohol syndrome (FAS), is preventable and remains a public health tragedy. The incidences of FAS and FASD have been conservatively estimated to be 0.97 and 10 per 1000 births, respectively. Meconium testing has been demonstrated to be a promising at-birth method for detection of drinking during pregnancy.
The current costs of FAS and FASD, alcohol treatment programs, and meconium screening were estimated by literature review. Monetary values were converted roughly to equal dollars in 2006.
Costs of adding meconium analysis to the current newborn screening program and of treatment for the identified mothers were estimated and compared to potential averted costs that may result from identification and intervention for mothers and affected infants. Three potential maternal treatment strategies are analyzed. Depending on the treatment type, the savings may range from 97 for every $1 spent on screening and treatment.
It needs to be emphasized, however, that such screening is premature and that to be effective this screening can be implemented only if there is a societal willingness to institute prevention and intervention programs to improve both women's and children's health. Future research should be directed at improving detection and developing in-depth prevention and remedial intervention programs. A thorough consideration of the ethical issues involved in such a screening program is also needed.
The main objective of this study is to evaluate the clinical utility of meconium analysis for fatty acid ethyl esters as a universal screening tool intended for the detection of newborns at risk for fetal alcohol spectrum disorder. This will be accomplished by assessing the rate of voluntary participation in a nonanonymous neonatal screening program and by determining the logistics of implementing the necessary follow-up and interventions as part of routine care. Additionally, this study will determine the predictive value of fatty acid ethyl ester-positive meconium with regard to neurodevelopmental delays. This is an ongoing prospective cohort study. Written informed consent is sought from all Grey Bruce women delivering at participating birthing sites. Collected meconium samples are tested for fatty acid ethyl esters by headspace-solid-phase microextraction followed by gas chromatography-mass spectrometry. Children with positive results are followed up through an existing public health program involving regular home visits and assessments of developmental milestones by a public health nurse. These children and matched control subjects also undergo neurodevelopmental testing at 3 and 18 months of age by a clinical psychologist using Bayley Scales of Infant and Toddler Development. If delays are detected, the child is referred to diagnostic services and appropriate intervention programs. This study has been granted ethics approval and enrollment began in November 2008 at St. Joseph's Health Care in London, Ontario. The first positive case has been identified and the follow-up is currently being conducted by the public health unit. The successful completing of this study will reveal the population's willingness to participate in a neonatal screening program for prenatal alcohol exposure and determine the costs, feasibility, and utility of implementing such programs in clinical practice.
To examine the conditions under which mothers would consent to alcohol and drug screening of their infants, and to identify predictors of screening consent.
A cross-sectional survey was administered in person by trained research assistants on the postpartum units of three hospitals in a large Canadian urban centre over four months. The survey was administered to 1509 mothers (78.4% of those eligible) who were fluent in English and had given birth within the preceding 48 hours.
Mothers indicated that they would consent to screening of their newborn (1369/1460, 93.8%), and thought all mothers should consent if infants at risk would be more likely to receive effective treatment (1440/1476, 97.6%). Respondents believed that they would consent to screening if they were provided the following information: what would happen if the infant sample was positive for prenatal exposure (1431/1476, 97%); who would have access to the information (1377/1476, 93.4%); how effective medical care would be for the child (1435/1476, 97.4%); and the likelihood that a baby with a positive screen would have a problem (1444/1476, 98.1%). Self-reported alcohol use did not decrease willingness to consent. In a multivariate model, belief that universal screening would not make women feel discriminated against was a significant predictor of consent (adjusted OR 5.9; 95% CI 3.3-10.6).
Mothers would support a universal newborn alcohol and drug screening program if there was evidence that screening could lead to effective treatment for the mother and baby, and if appropriate resources were available.
Very rarely therapeutic drug monitoring is evaluated for its ability to provide cost savings. A new article describes the cost-effectiveness of meconium analysis for fatty acid ethyl esters in the management of fetal alcohol spectrum disorder. This article is a summary and a critical appraisal of the article by Hopkins et al.
Fetal alcohol spectrum disorder (FASD) is the most common cause of neurobehavioural handicap in North America. Screening for FASD may facilitate diagnosis and hence management of these children. We present a variety of screening tools for the identification of children at risk for FASD.
We critically reviewed and evaluated published and practiced methods for their potential of screening suspected cases, their epidemiological characteristics (sensitivity, specificity, positive and negative predictive values) [Phase I], as well as their feasibility [Phase II].
The following five tools were selected for the FASD screening toolkit: screening fatty acid ethyl esters in neonatal meconium, the modified Child Behaviour Checklist, Medicine Wheel tool, Asante Centre Probation Officer Tool, and maternal history of drinking and drug use.
The toolkit for FASD screening aims at screening different populations, from the newborns to youth and at-risk mothers. It is anticipated that the toolkit will facilitate diagnosis of FASD.
The diagnosis of fetal alcohol syndrome (FAS) was invented in 1973. This paper investigates the process by which a cluster of birth defects associated with exposure to alcohol in utero came to be a distinct medical diagnosis, focusing on the first ten years of the medical literature on FAS. Fetal alcohol syndrome was "discovered" by a group of American dysmorphologists who published the first case reports and coined the term FAS. However, the nature of the diagnosis and its salient symptoms were determined collectively over time by the medical profession as a whole. The paper traces the natural history of the diagnosis in the U.S. through five stages: introduction, confirmation and corroboration, dissent, expansion, and diffusion. FAS serves as an example of the social construction of clinical diagnosis; moral entrepreneurship plays a key role and the medical literature on FAS is infused with moral rhetoric, including passages from classical mythology, philosophy, and the Bible. FAS is a moral as well as a medical diagnosis, reflecting the broader cultural concerns of the era in which it was discovered, including a greater awareness of environmental threats to health, the development of fetal medicine, an emphasis on "the perfect child," and a growing paradigm of maternal-fetal conflict.
National statistics indicate that 11% of newborns are exposed to drugs of abuse in utero (Chasnoff, Burns, Schnoll, & Burns, 1985). From July through November of 1993, 0.2% (1/583) of infants in our hospital screened positive for exposure to drugs of abuse in utero, by blood and urine screens collected per physician's order. During the same 4 months in 1995, 0.2% (1/574) screened positive. In contrast, from July through November 1994, all newborns born at our institution were screened for in utero drug exposure using meconium samples; approximately 4.5% (22/493) were positive. Meconium screening of all infants, not just those per physician order, produced a dramatic increase in positive drug results. Of great concern in this study was that only two of the 22 mothers identified as having drug-screen positive babies during the 1994 meconium screening agreed to use rehabilitative services. This highlights the necessity of intervention through early educational programs stressing prevention.
According to the U.S. Supreme Court, the Fourth Amendment rights of 10 women were violated by a hospital that provided them prenatal care. The incidence of prenatal drug testing for criminal prosecution with or without a woman's knowledge is increasing. Concurrently, funding and availability of drug treatment programs for pregnant women are declining. Nurses and physicians who act as advocates for the state rather than the patient damage the patient-provider relationship and breach their ethical responsibility to the patient.
We sought to determine the attitudes of obstetricians, pediatricians, and family practice physicians in Michigan concerning involvement of the criminal justice system in preventing drug and alcohol abuse during pregnancy.
Physicians were sent a questionnaire by mail asking for their agreement with statements concerning the involvement of the criminal justice system with respect to substance abuse during pregnancy.
Nearly all (95%) agreed that pregnant women have a moral duty to ensure they had healthy babies; 59% agreed that they should also have a legal responsibility to do so. Most physicians (77%) agreed that screening for acquired immunodeficiency syndrome during pregnancy should be mandatory. Almost as high a percentage (61% to 75% depending on subspecialty) were also in favor of mandatory screening for alcohol abuse; agreement for screening for illicit drugs was much lower (43% to 55% depending on subspecialty). Despite their consensus (61%) that fear of prosecution would deter pregnant abusers from seeking prenatal care, most were in agreement that existing laws regarding child abuse and neglect need to be redefined to include alcohol (54%) and drug abuse (61%) during pregnancy; 52% were in favor of enacting a statute that includes drug or alcohol use during pregnancy as "child abuse" for purposes of removing that child from maternal custody. Physicians were highly in favor of compulsory treatment for illicit drug use and alcohol abuse for women already in the criminal justice system (82%-83%), neutral with respect to court-ordered contraception for alcohol- (50%) and drug-abusing women (47%), and opposed to criminal prosecution for either alcohol abuse (18%-31% depending on subspecialty) or illicit drug use (23%-34%) during pregnancy.
Other than criminal prosecution, physicians are not opposed to involvement of the legal justice system in preventing alcohol and drug abuse during pregnancy.
Clinical descriptions of patients with Fetal Alcohol Syndrome (FAS) and Fetal Alcohol Effects (FAE) suggest major problems with adaptive behavior. Five operationally defined adverse outcomes and 18 associated risk/protective factors were examined using a Life History Interview with knowledgeable informants of 415 patients with FAS or FAE (median age 14 years, range 6-51; median IQ 86, range 29-126). Eighty percent of these patients were not raised by their biological mothers. For adolescents and adults, the life span prevalence was 61% for Disrupted School Experiences, 60% for Trouble with the Law, 50% for Confinement (in detention, jail, prison, or a psychiatric or alcohol/drug inpatient setting), 49% for Inappropriate Sexual Behaviors on repeated occasions, and 35% for Alcohol/Drug Problems. The odds of escaping these adverse life outcomes are increased 2- to 4-fold by receiving the diagnosis of FAS or FAE at an earlier age and by being reared in good stable environments.
The diagnosis of fetal alcohol spectrum disorder is a difficult task, especially in cases where clear, physical markers of in utero alcohol exposure are not apparent. Reviewed in the following paper are some older tools for screening alcohol use in pregnancy and present novel approaches to the diagnosis of FASD, including ethanol biomarker development to behavioural phenotyping. Improving current FASD diagnostic methodology through more novel approaches may provide the possibility of earlier and wider diagnosis, allowing intervention and treatment at stages where the advanced effects of alcohol can still be mitigated.
Researchers have been actively looking to biomarker development as a way to improve diagnosis in conditions such as fetal alcohol syndrome (FAS) that have typically been difficult to identify at an early stage. Meconium testing is considered a potentially useful newborn screening method. Screening for alcohol (and other drug) use is unique from all other types of newborn screening in that there is a greater element of social risk for parents, particularly mothers (public exposure of substance use with potential for child welfare involvement). There are many factors related to the science and ethics of the meconium screening process to consider before implementing universal or targeted screening. As care providers who participate in the screening and counseling process and as advocates for infants and their families, neonatal nurses should be active participants in discussions surrounding the ethical and clinical appropriateness of meconium screening program development and expansion. The science behind meconium screening at present is not strong enough to warrant widespread implementation of screening; neonatal nurses are cautioned to approach screening carefully because of the critical social implications for mother and baby.
While newborn blood spot screening has historically been viewed as a public health success, the potential harms and benefits are more finely balanced for new conditions being considered for program expansion. We highlight complex issues that must be addressed in policy decisions, which in turn requires a consideration of many stakeholder perspectives. Using national policy documents from the United Kingdom, the United States, Australia, and Canada, we describe the participation of stakeholder organizations in the newborn screening policy process, how such organizations have incorporated stakeholder views into their own policy writing, and their recommendations for inclusiveness. Stakeholder participation in newborn screening decision-making is widely acknowledged as important, and many methods have been endorsed - consultation as well as direct or indirect input into policy development. Differences across organizations and jurisdictions raise questions about the most effective approaches for facilitating inclusiveness, suggesting a need for formal evaluative research.
Challenges in identifying children exposed prenatally to ethanol necessitate the development of a biomarker for neonates at risk for fetal alcohol spectrum disorder. Meconium fatty acid ethyl esters (FAEE), products of nonoxidative ethanol metabolism, have been established as a novel biomarker of fetal ethanol exposure. We present the first application of this biomarker to a population-based sample in Canada. Six-hundred eighty-two meconium specimens were anonymously collected in the region of Grey Bruce, Ontario, Canada. Meconium FAEE were extracted by liquid-liquid and solid-phase extraction and analyzed by gas chromatography with flame-ionization detection confirmed by gas chromatography with mass spectrometry. We measured ethyl palmitate (E16:0), ethyl palmitoleate (E16:1), ethyl stearate (E18:0), ethyl oleate (E18:1), ethyl linoleate (E18:2), ethyl linolenate (E18:3), and ethyl arachidonate (E20:4). Seventeen of 682 meconium samples tested positive for significant prenatal ethanol exposure (>2.0 nmol/g). FAEE analysis detected fivefold more ethanol-exposed pregnancies than standard postpartum questionnaires in this population (2.5% versus 0.5%) (P < 0.001). The prevalence of ethanol-exposed pregnancies was consistent with Centers for Disease Control and Prevention estimates of "frequent" prenatal drinking and previously published estimates of fetal alcohol spectrum disorder disease prevalence in the general North American population. The FAEE concentrations of negative (95% confidence interval, 0.38-0.49 nmol/g) versus positive (95% confidence interval, 7.74-151.28 nmol/g) samples were distinct, further demonstrating the specificity of this biomarker in determining significant prenatal ethanol exposure. Meconium FAEE analysis demonstrates a fivefold increase in sensitivity over currently used methods of self-report-based screening in Ontario for the detection of ethanol-exposed pregnancies in a clinical setting.
Meconium alcohol and drug screening University of Calgary Calgary
- M Hicks
Hicks M (2007) Meconium alcohol and drug screening. PhD Thesis, University of Calgary, Calgary.