Background:Fetal alcohol spectrum disorders (FASD) affect 1.1% to 5% of the general population. Yet, most children with FASD and their families cannot access evidence-based interventions. Mobile health (mHealth) interventions have the potential to increase access to care on a broad scale. While numerous self-directed parenting apps exist, none have been tested for FASD. The FMF (Families Moving Forward) Connect app is a self-directed intervention derived from an empirically supported intervention for caregivers raising children with FASD. FMF Connect is the first self-directed parenting app for FASD, and also one of the first parenting apps to be systematically developed and tested. Objective:This study aimed to test the efficacy of FMF Connect for caregivers raising children with FASD on targeted primary (child behavior, caregiver attributions, parenting efficacy and satisfaction, FASD knowledge, and family needs met) and secondary (child adaptive behavior, caregiver self-care, and app satisfaction) outcomes. Methods:This study involved a 3-arm randomized controlled trial with equal allocation to groups (1) FMF Connect+coaching, (2) FMF Connect, or (3) waitlist control. Participants from the United States were recruited online through an open access website. Recruitment materials were distributed by the Collaborative Initiative on FASD, FASD listserves, and social media. In total, 129 caregivers of children (aged 3-12 y) with FASD or prenatal alcohol exposure (PAE) were enrolled. Online surveys were administered at baseline, 6 weeks, and 12 weeks. Data were analyzed with linear mixed modeling, linear regressions, and structural equation modeling using SPSS (version 29.0; IBM) and Mplus 8 (Muthén & Muthén). Results:A total of 43 participants were randomized to each group. Caregivers were predominantly White adoptive mothers. Of the total, 64% (n=83) of participants were retained through the 12-week follow-up. Groups did not differ in terms of demographic characteristics, baseline levels of functioning, or attrition. Usage patterns were similar across groups, suggesting coaching did not increase engagement. Given a few differences, app intervention groups were combined for analyses. Relative to the waitlist group, caregivers in the FMF Connect group evidenced greater improvements in FASD knowledge, child behavior attributions, family needs met, and self-care after 12 weeks (P=.01-.048). After controlling for multiple comparisons, differences in FASD knowledge, self-care, and family needs met approached significance (P=.06-.07). Groups did not differ in parenting satisfaction, child behavior problems, or adaptive functioning. More app usage is related to greater changes in parenting efficacy. Caregiver behavior attributions at 6 weeks did not mediate intervention effects. Conclusions:This study demonstrated initial efficacy of the FMF Connect app for targeted caregiver outcomes, with small to medium effect sizes. As an mHealth app, the FMF Connect intervention has potential for scalability and accessibility. This could lead to a substantial public health impact, particularly for families who face challenges accessing evidence-based resources or encounter other barriers to care.

Background:An estimated 1 in 4 children seeking mental health care have an undiagnosed fetal alcohol spectrum disorder (FASD). Few mental health providers receive training or feel competent to serve this population. Evidence-based interventions (EBIs) for FASD have low uptake across mental health settings. Objective:This study aimed to systematically adapt an EBI to increase access to care for youth with FASD in mental healthcare settings, resulting in the innovative Families Moving Forward (FMF) Connect Pro program. Method:A multidisciplinary planning group employed the theoretically-driven implementation science framework, Intervention Mapping-Adapt (IM-Adapt). This 6-step process incorporated multiple information sources, including literature review, theory, clinical wisdom, and focus groups. 18 community mental health providers participated in focus groups to assess the fit of the adapted program. Data were analyzed with rapid qualitative methods using the COM-B model (Capability, Opportunity, Motivation) of behavior change and Theoretical Domains Framework (TDF) for coding and interpretation. Results:Step 1: Planning group developed a logic model of change. Step 2: Review of EBIs, selecting the Families Moving Forward (FMF) intervention and two training methods: Extension of Community Healthcare Outcomes (ECHO) and Self-Directed modular approaches. Steps 3&4: Planning group identified needed adaptations for providers with low utilization of FASD-informed care and flexible use in diverse settings. Focus group participants reviewed prototypes of the resulting FMF Connect Pro program, endorsing high acceptability. TDF determinants across domains of capability, opportunity, and motivation were highlighted as contributing factors to provider behavior change. Step 5: Implementation/evaluation plans were developed. Conclusions:Results reveal the FMF Connect Pro program was deemed acceptable and appears a promising method to expand FASD-informed care. A clinical trial is now underway (Step 6). Additionally, this study models an IM-Adapt approach that could be applied to increase broader adoption and implementation of EBIs.

BACKGROUND:Students with fetal alcohol spectrum disorders (FASD) are at risk of poor academic performance and school disruption given teachers' limited opportunity to learn about how to support them. The Families Moving Forward (FMF) Program and its derivative mobile health intervention, FMF Connect, provide an ideal framework to inform a scalable intervention for teachers. The current study aimed to develop and test the FMF Connect Teacher Companion website using the ADAPT-ITT framework, including teacher input. METHODS:With teacher input, materials from the FMF Program and FMF Connect were adapted to be relevant to teachers' needs and the classroom setting. Next, 37 teachers participated in a fully remote, nonblinded, randomized two-group parallel design pilot trial of the website. Eligible teachers had direct contact with a student with FASD aged 5-12 in an educational setting. The pilot trial was preregistered and publicly available on ClinicalTrials.gov (study no.: NCT05986565) prior to recruitment commencement. Descriptive statistical analysis aimed to understand technological functionality and study design acceptability. RESULTS:Findings of both website (n = 21) and control (n = 16) groups demonstrated the website was satisfactory and functional. Participants had positive impressions of all content but reported some negative impressions around aesthetics and the large amount of information. CONCLUSIONS:The FMF Connect Teacher Companion website is a feasible, acceptable FASD-informed resource for teachers.

BACKGROUND:Recent interest in the screening and diagnosis of fetal alcohol spectrum disorders (FASD) by the American Psychiatric Association and the American Association of Pediatrics has highlighted the lack of empirical evidence regarding the levels and patterns of prenatal alcohol exposure (PAE) associated with clinically significant adverse effects on cognitive and behavioral function. We used benchmark dose analysis to address this issue. METHODS:Benchmark dose analyses were performed on composite cognitive function scores derived from data obtained at school age, adolescence, and early adulthood from 2227 participants from six prospective, longitudinal cohorts, whose mothers were interviewed about their alcohol and drug use during pregnancy or shortly thereafter. We compared single-predictor models based on average alcohol consumed per day during pregnancy with a two-predictor, semiparametric model based on (1) average alcohol dose per drinking occasion and (2) drinking frequency. RESULTS:Our two-predictor model showed that at lower levels of drinking frequency, a relatively high dose/occasion is required for an increased risk of poor intellectual function, whereas at higher levels of drinking frequency, a lower dose/occasion is sufficient to increase that risk. In addition, lower doses/occasion were associated with clinically meaningful adverse effects in participants born to older than to younger mothers. The single-predictor models proved less adequate because the dose/occasion for a substantial proportion of participants exposed at the benchmark doses generated by those models was too low to increase the risk of clinically significant effects. CONCLUSIONS:Given that the cognitive and behavioral deficits seen in FASD resemble those also seen in a range of other disorders, whether the patient's PAE is sufficient to increase the risk of clinically meaningful impairment is a critical element in an FASD diagnosis. Accurate diagnosis is needed to determine which patients warrant interventions that have been shown to be effective in remediating PAE-related neurobehavioral impairment.

While benchmark dose (BMD) methodology is well-established for settings with a single exposure, these methods cannot easily handle multidimensional exposures with nonlinear effects. We propose a framework for BMD analysis to characterize the joint effect of a two-dimensional exposure on a continuous outcome using a generalized additive model while adjusting for potential confounders via propensity scores. This leads to a dose-response surface which can be summarized in two dimensions by a contour plot in which combinations of exposures leading to the same expected effect are identified. In our motivating study of prenatal alcohol exposure, cognitive deficits in children are found to be associated with both the frequency of drinking as well as the amount of alcohol consumed on each drinking day during pregnancy. The general methodological framework is useful for a broad range of settings, including combinations of environmental stressors, such as chemical mixtures, and in explorations of the impact of dose rate rather than simply cumulative exposure on adverse outcomes.

BACKGROUND:Most studies of the effects of prenatal alcohol exposure (PAE) on cognitive function have assumed that the dose-response curve is linear. However, data from a few animal and human studies suggest that there may be an inflection point in the dose-response curve above which PAE effects are markedly stronger and that there may be differences associated with pattern of exposure, assessed in terms of alcohol dose per drinking occasion and drinking frequency.METHODS:We performed second-order confirmatory factor analysis on data obtained at school age, adolescence, and early adulthood from 2227 participants in six US longitudinal cohorts to derive a composite measure of cognitive function. Regression models were constructed to examine effects of PAE on cognitive function, adjusted for propensity scores. Analyses based on a single predictor (absolute alcohol (AA)/day) were compared with analyses based on two predictors (dose/occasion and drinking frequency), using (1) linear models and (2) nonparametric general additive models (GAM) that allow for both linear and nonlinear effects.RESULTS:The single-predictor GAM model showed virtually no nonlinearity in the effect of AA/day on cognitive function. However, the two-predictor GAM model revealed differential effects of maternal drinking pattern. Among offspring of infrequent drinkers, PAE effects on cognitive function were markedly stronger in those whose mothers drank more than ~3 drinks/occasion, and the effect of dose/occasion was strongest among the very frequent drinkers. Frequency of drinking did not appear to alter the PAE effect on cognitive function among participants born to mothers who limited their drinking to ~1 drink/occasion or less.CONCLUSIONS:These findings suggest that linear models based on total AA/day are appropriate for assessing whether PAE affects a given cognitive outcome. However, examination of alcohol dose/occasion and drinking frequency is needed to fully characterize the impact of different levels of alcohol intake on cognitive impairment.

High levels of prenatal alcohol exposure (PAE) result in significant cognitive deficits in children, but the exact nature of the dose-response relationship is less well understood. To investigate this relationship, data were assembled from six longitudinal birth cohort studies examining the effects of PAE on cognitive outcomes from early school age through adolescence. Structural equation models (SEMs) are a natural approach to consider, because of the way they conceptualise multiple observed outcomes as relating to an underlying latent variable of interest, which can then be modelled as a function of exposure and other predictors of interest. However, conventional SEMs could not be fitted in this context because slightly different outcome measures were used in the six studies. In this paper we propose a multi-group Bayesian SEM that maps the unobserved cognition variable to a broad range of observed outcomes. The relation between these variables and PAE is then examined while controlling for potential confounders via propensity score adjustment. By examining different possible dose-response functions, the proposed framework is used to investigate whether there is a threshold PAE level that results in minimal cognitive deficit.

BACKGROUND:Children with fetal alcohol spectrum disorders (FASD) have high rates of adverse childhood experiences (ACEs). ACEs are associated with a wide range of health outcomes including difficulty with behavior regulation, an important intervention target. However, the effect of ACEs on different areas of behavior has not been well characterized in children with disabilities. This study describes ACEs in children with FASD and how they impact behavior problems.METHODS:A convenience sample of 87 caregivers of children (aged 3 to 12) with FASD participating in an intervention study reported on their children's ACEs using the ACEs Questionnaire and behavior problems on the Eyberg Child Behavior Inventory (ECBI). A theorized three-factor structure of the ECBI (Oppositional Behavior, Attention Problems, and Conduct Problems) was investigated. Data were analyzed using Pearson correlations and linear regression.RESULTS:On average, caregivers endorsed 3.10 (SD = 2.99) ACEs experienced by their children. The two most frequently endorsed ACE risk factors were having lived with a household member with a mental health disorder, followed by having lived with a household member with a substance use disorder. Higher total ACEs score significantly predicted a greater overall frequency of child behavior (intensity scale), but not whether the caregiver perceived the behavior to be a problem (problem scale) on the ECBI. No other variable significantly predicted the frequency of children's disruptive behavior. Exploratory regressions indicated that a higher ACEs score significantly predicted greater Conduct Problems. Total ACEs score was not associated with Attention Problems or Oppositional Behavior.DISCUSSION:Children with FASD are at risk for ACEs, and those with higher ACEs had a greater frequency of problem behavior on the ECBI, especially conduct problems. Findings emphasize the need for trauma-informed clinical care for children with FASD and increased accessibility of care. Future research should examine potential mechanisms that underlie the relationship between ACEs and behavior problems to optimally inform interventions.

Evidence from animal models and epidemiological studies has linked prenatal alcohol exposure (PAE) to a broad range of long-term cognitive and behavioral deficits. However, there is virtually no information in the scientific literature regarding the levels of PAE associated with an increased risk of clinically significant adverse effects. During the period from 1975-1993, several prospective longitudinal cohort studies were conducted in the U.S., in which maternal reports regarding alcohol use were obtained during pregnancy and the cognitive development of the offspring was assessed from early childhood through early adulthood. The sample sizes in these cohorts did not provide sufficient power to examine effects associated with different levels and patterns of PAE. To address this critical public health issue, we have developed a hierarchical meta-analysis to synthesize information regarding the effects of PAE on cognition, integrating data on multiple endpoints from six U.S. longitudinal cohort studies. Our approach involves estimating the dose-response coefficients for each endpoint and then pooling these correlated dose-response coefficients to obtain an estimated `global' effect of exposure on cognition. In the first stage, we use individual participant data to derive estimates of the effects of PAE by fitting regression models that adjust for potential confounding variables using propensity scores. The correlation matrix characterizing the dependence between the endpoint-specific dose-response coefficients estimated within each cohort is then run, while accommodating incomplete information on some endpoints. We also compare and discuss inferences based on the proposed approach to inferences based on a full multivariate analysis

Purpose of the Review The current review proposes a theoretical framework to support professionals in collaborating with families in the provision of services for children with fetal alcohol spectrum disorders (FASD). Existing models of family-directed care and family contextual factors relevant to planning interventions were reviewed. This information was adapted and integrated in the context of available evidence regarding the provision of evidence-based approaches for children with FASD and their families. Recent Findings The proposed theoretical framework integrates a family-directed approach to brain injury model, which includes key components of hope, family expertise, and education/skill building with the social economy model of excluded families. This provides a comprehensive approach to supporting the complex needs of children with FASD and their families. Conclusions Specialist services for children with FASD and their families are significantly limited around the world. The proposed model provides a theoretical framework for educating and supporting practitioners and family members, to facilitate collaborative service provision for children with FASD and their families.

BACKGROUND:Fetal alcohol spectrum disorders (FASD) are prevalent neurodevelopmental conditions. Significant barriers prevent family access to FASD-informed care. To improve accessibility, a scalable mobile health intervention for caregivers of children with FASD is under development. The app, called Families Moving Forward (FMF) Connect, is derived from the FMF Program, a parenting intervention tailored for FASD. FMF Connect has 5 components: Learning Modules, Family Forum, Library, Notebook, and Dashboard.OBJECTIVE:This study assesses the feasibility of FMF Connect intervention prototypes. This includes examining app usage data and evaluating user experience to guide further refinements.METHODS:Two rounds of beta-testing were conducted as part of a systematic approach to the development and evaluation of FMF Connect: (1) an iOS prototype was tested with 20 caregivers of children (aged 3-17 years) with FASD and 17 providers for the first round (April-May 2019) and (2) iOS and Android prototypes were tested with 25 caregivers and 1 provider for the second round (November-December 2019). After each 6-week trial, focus groups or individual interviews were completed. Usage analytics and thematic analysis were used to address feasibility objectives.RESULTS:Across beta-test trials, 84% (38/45) of caregivers and 94% (17/18) of providers installed the FMF Connect app. Technological issues were tracked in real time with updates to address problems and expand app functionalities. On use days, caregivers averaged 20 minutes using the app; most of the time was spent watching videos in Learning Modules. Caregiver engagement with the Learning Modules varied across 5 usage pattern tiers. Overall, 67% (30/45) of caregivers posted at least once in the Family Forum. Interviews were completed by 26 caregivers and 16 providers. App evaluations generally did not differ according to usage pattern tier or demographic characteristics. Globally, app users were very positive, with 2.5 times more positive- than negative-coded segments across participants. Positive evaluations emphasized the benefits of accessible information and practical utility of the app. Informational and video content were described as especially valuable to caregivers. A number of affective and social benefits of the app were identified, aligning well with the caregivers' stated motivators for app use. Negative evaluations of user experience generally emphasized technical and navigational aspects. Refinements were made on the basis of feedback during the first beta test, which were positively received during the second round. Participants offered many valuable recommendations for continuing app refinement, which is useful in improving user experience.CONCLUSIONS:The results demonstrate that the FMF Connect intervention is acceptable and feasible for caregivers raising children with FASD. They will guide subsequent app refinement before large-scale randomized testing. This study used a systematic, user-centered design approach for app development and evaluation. The approach used here may illustrate a model that can broadly inform the development of mobile health and digital parenting interventions.

Individuals with fetal alcohol spectrum disorders (FASD) often contend with daily life challenges. For many years, these have been called "secondary disabilities." Ranging widely, these are described as school disruption, legal problems, independent living needs, mental health difficulties, and more. Starting in the mid-1990's, in multiple countries, clinic-based and national register studies documented elevated rates of problems in adaptive behavior and secondary disabilities occurring for individuals with FASD (e.g., Streissguth et al., 1996; Streissguth et al., 2004; Spohr et al., 2007; Temple et al., 2011; Rangmar et al., 2015). Accumulating data revealed these challenges as ongoing, functionally impairing, and especially troubling as individuals with FASD advanced into adolescence and adulthood.

Background Cognitive and behavioral sequelae of prenatal alcohol exposure (PAE) continue to be prevalent in the United States and worldwide. Because these sequelae are also common in other neurodevelopmental disorders, researchers have attempted to identify a distinct neurobehavioral profile to facilitate the differential diagnosis of fetal alcohol spectrum disorders (FASD). We used an innovative, individual participant meta-analytic technique to combine data from six large U.S. longitudinal cohorts to provide a more comprehensive and reliable characterization of the neurobehavioral deficits seen in FASD than can be obtained from smaller samples. Methods Meta-analyses were performed on data from 2236 participants to examine effects of PAE (measured as oz absolute alcohol/day (AA/day)) on IQ, four domains of cognition function (learning and memory, executive function, reading achievement, and math achievement), sustained attention, and behavior problems, after adjusting for potential confounders using propensity scores. Results The effect sizes for IQ and the four domains of cognitive function were strikingly similar to one another and did not differ at school age, adolescence, or young adulthood. Effect sizes were smaller in the more middle-class Seattle cohort and larger in the three cohorts that obtained more detailed and comprehensive assessments of AA/day. PAE effect sizes were somewhat weaker for parent- and teacher-reported behavior problems and not significant for sustained attention. In a meta-analysis of five aspects of executive function, the strongest effect was on set-shifting. Conclusions The similarity in the effect sizes for the four domains of cognitive function suggests that PAE affects an underlying component or components of cognition involving learning and memory and executive function that are reflected in IQ and academic achievement scores. The weaker effects in the more middle-class cohort may reflect a more cognitively stimulating environment, a different maternal drinking pattern (lower alcohol dose/occasion), and/or better maternal prenatal nutrition. These findings identify two domains of cognition-learning/memory and set-shifting-that are particularly affected by PAE, and one, sustained attention, which is apparently spared.

Date Presented 03/28/20 This qualitative study explored the converging and diverging needs of biological and adoptive parents raising young children with or at risk for fetal alcohol spectrum disorders. Findings from this study advocate for early interventions that prioritize and promote social-emotional development of young children, are family-centered, and provide concrete assistance for family circumstances and parent well-being. Primary Author and Speaker: Misty Pruner Contributing Authors: Tracy Jirikowic, Kathryn Yorkston, Heather Carmichael Olson

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

While it is well known that high levels of prenatal alcohol exposure (PAE) result in significant cognitive deficits in children, the exact nature of the dose response is less well understood. In particular, there is a pressing need to identify the levels of PAE associated with an increased risk of clinically significant adverse effects. To address this issue, data have been combined from six longitudinal birth cohort studies in the United States that assessed the effects of PAE on cognitive outcomes measured from early school age through adolescence. Structural equation models (SEMs) are commonly used to capture the association among multiple observed outcomes in order to characterise the underlying variable of interest (in this case, cognition) and then relate it to PAE. However, it was not possible to apply classic SEM software in our context because different outcomes were measured in the six studies. In this paper we show how a Bayesian approach can be used to fit a multi-group multi-level structural model that maps cognition to a broad range of observed variables measured at multiple ages. These variables map to several different cognitive subdomains and are examined in relation to PAE after adjusting for confounding using propensity scores. The model also tests the possibility of a change point in the dose-response function.

Background: Fetal alcohol spectrum disorders (FASD) affect approximately 2% to 5% of the US population. However, most families are unable to access FASD-informed interventions. Barriers to care include the lack of a knowledgeable and skilled workforce and family-level barriers such as limited financial resources, inability to access childcare, and stigma. As a result, families often try peer-to-peer and self-help support strategies. However, they often take these strategies from disparate sources, which have quite variable intervention quality and empirical support. Objective: This study aimed to initiate systematic development and evaluation of a mobile health intervention (app) for caregivers raising children with FASD. Focus groups were conducted to elicit participant perspectives on app design and functionalities to inform further app development. Methods: The app, called FMF Connect, was derived from the scientifically validated Families Moving Forward (FMF) Program, a clinician-delivered behavioral consultation intervention. FMF Connect was intended for caregiver self-delivery and included five main components: (1) Learning Modules, (2) Family Forum, (3) Library, (4) Notebook, and (5) Dashboard. Focus group methods were used to solicit perspectives from diverse families during the early stages of app development. Questions were asked about interface design, relevance of components and content, and perceived barriers and facilitators of use. A total of 25 caregivers participated in 7 focus groups across 5 US cities. Data were analyzed thematically. Results: Focus group participants were generally enthusiastic about the app interface design and components. Four global positive impression themes emerged, including (1) ease of access, (2) how the app guides and organizes information, (3) connection to other users and information, and (4) ability to share some content with others. Themes arose not only in discussions relating to positive app features but also when participants were asked about motivators for app use. Participants related how these positive global themes could address some system-level barriers, such as limited access to services, feeling isolated, and increased advocacy needs related to the societal lack of FASD knowledge. Participants identified many positive features about individual app components and functionalities. They also communicated potential barriers to use and raised important concerns and considerations relating to several app components. These included recognizability of the app based on the logo, and the balance of following the planned intervention sequence versus obtaining immediate answers. Also mentioned were privacy and dynamics within the Family Forum. Conclusions: FMF Connect is a promising novel intervention with potential to reach many families in need and reduce significant barriers to care, resulting in a broader public health impact. Study findings will guide further app development both in terms of content and technological advances to optimize intervention effects. FMF Connect app development provides useful directions for other apps aimed at changing parenting practices.