Experiences of Children with Fetal Alcohol Spectrum Disorder and Their Families: A Critical Review.

Evidence suggests that children with fetal alcohol spectrum disorder (FASD) experience challenges across many areas of their daily lives and often require interprofessional supports. Recent studies have emphasized the need for an integrated system of care for children with FASD, incorporating medical, allied health, and education services, to facilitate open communication and support for the complex needs that many children experience. To develop such a system of care, it is important to first understand the impact of FASD on children's functioning during daily activities in different environmental contexts. A critical review of existing research was conducted using a critical interpretive synthesis approach. Results revealed that while many studies discussed impacts at the body functions and structures level of children with FASD, they often did not consider the activity, participation, and environmental factors also contributing to the daily functioning of this population. Several studies discussed caregiver experiences and challenges raising a child with FASD; however, no studies investigated the lived experiences relating to impacts across activities and environments from children's perspectives. In addition, the focus on deficits overshadowed investigation into the strengths of children with FASD, leaving a gap in the picture of their daily lives. Further research is required to determine the strengths that children with FASD demonstrate and the challenges impacting their daily functioning within different environmental contexts. Insights gleaned from such research would support intervention practices to become more holistic and interprofessional.

FAST Club: The Impact of a Physical Activity Intervention on Executive Function in Children With Fetal Alcohol Spectrum Disorder.

Physical activity (PA) has been demonstrated to have positive effects on cognitive function, particularly executive function (EF) skills. Animal models suggest PA may be effective in ameliorating some of the neuropsychological effects of fetal alcohol spectrum disorder (FASD), but this approach has not been extended to humans. The purpose of this study was to develop a PA program, FAST Club, for children with FASD and to evaluate its effect on a measure of EF. Using a wait-list control design, 30 children age 7-14 yr participated in FAST Club for 2 × 1.5-hr sessions/week for 8 weeks. EF was assessed using the Children's Color Trails Test. Significant improvements in T scores on the Children's Color Trails Test were seen immediately postprogram, and this improvement was sustained at 3 months postprogram. These findings provide evidence to support the use of PA as a means to improve EF in children with FASD.

The development of a culturally appropriate school based intervention for Australian Aboriginal children living in remote communities: A formative evaluation of the Alert Program® intervention.

BACKGROUND/AIM: Although previous research has demonstrated the benefits of targeting self-regulation in non-Aboriginal children, it is unclear whether such programs would be effective for Aboriginal children attending school in remote communities. Some of these children have been diagnosed with a fetal alcohol spectrum disorder (FASD) impairing their ability to self-regulate. The aim of this article is to describe a three phase formative process to develop and pilot a curriculum version of the Alert Program® , a promising intervention for improving self-regulation that could be used in remote community schools. This modified version of the program will be subsequently tested in a cluster randomised controlled trial. METHODS: A mixed methods approach was used. RESULTS: Modifications to the Alert Program® , its delivery and evaluation were made after community and stakeholder consultation facilitated by a senior Aboriginal community researcher. Changes to lesson plans and program resources were made to reflect the remote community context, classroom environment and the challenging behaviours of children. Standardised study outcome measures were modified by removing several questions that had little relevance to the lives of children in remote communities. Program training for school staff was reduced in length to reduce staff burden. CONCLUSIONS: This study identified aspects of the Alert Program® training, delivery and measures for evaluation that need modification before their use in assessing the efficacy of the Alert Program® in remote Aboriginal community primary schools.

Interventions to improve gross motor performance in children with neurodevelopmental disorders: a meta-analysis.

BACKGROUND: Gross motor skills are fundamental to childhood development. The effectiveness of current physical therapy options for children with mild to moderate gross motor disorders is unknown. The aim of this study was to systematically review the literature to investigate the effectiveness of conservative interventions to improve gross motor performance in children with a range of neurodevelopmental disorders. METHODS: A systematic review with meta-analysis was conducted. MEDLINE, EMBASE, AMED, CINAHL, PsycINFO, PEDro, Cochrane Collaboration, Google Scholar databases and clinical trial registries were searched. Published randomised controlled trials including children 3 to ≤18 years with (i) Developmental Coordination Disorder (DCD) or Cerebral Palsy (CP) (Gross Motor Function Classification System Level 1) or Developmental Delay or Minimal Acquired Brain Injury or Prematurity (<30 weeks gestational age) or Fetal Alcohol Spectrum Disorders; and (ii) receiving non-pharmacological or non-surgical interventions from a health professional and (iii) gross motor outcomes obtained using a standardised assessment tool. Meta-analysis was performed to determine the pooled effect of intervention on gross motor function. Methodological quality and strength of meta-analysis recommendations were evaluated using PEDro and the GRADE approach respectively. RESULTS: Of 2513 papers, 9 met inclusion criteria including children with CP (n = 2) or DCD (n = 7) receiving 11 different interventions. Only two of 9 trials showed an effect for treatment. Using the least conservative trial outcomes a large beneficial effect of intervention was shown (SMD:-0.8; 95% CI:-1.1 to -0.5) with "very low quality" GRADE ratings. Using the most conservative trial outcomes there is no treatment effect (SMD:-0.1; 95% CI:-0.3 to 0.2) with "low quality" GRADE ratings. Study limitations included the small number and poor quality of the available trials. CONCLUSION: Although we found that some interventions with a task-orientated framework can improve gross motor outcomes in children with DCD or CP, these findings are limited by the very low quality of the available evidence. High quality intervention trials are urgently needed.

Virtual Sensorimotor Training for Balance: Pilot Study Results for Children With Fetal Alcohol Spectrum Disorders.

PURPOSE: To examine the effects of Sensorimotor Training to Affect Balance, Engagement, and Learning (STABEL), a virtual reality system to train sensory adaptation for balance control, for children with fetal alcohol spectrum disorders (FASDs). METHODS: Twenty-three children with FASDs received STABEL training in a university laboratory, or home, or were controls. The Movement Assessment Battery for Children-2nd edition (MABC-2) and Pediatric Clinical Test of Sensory Interaction for Balance-2 (P-CTSIB-2) were analyzed by group (lab, home, and control), session (pre-STABEL, 1 week post-STABEL, and 1 month post-STABEL), and group-by-session interaction. RESULTS: Significant effects were group and session for MABC-2 Balance and interaction for MABC-2 Total Motor and P-CTSIB-2. CONCLUSION: Preliminary results support improved sensory adaptation, balance, and motor performance post-STABEL, which warrant further study with a larger, randomized sample.

Positive behavior support (PBS) with children with fetal alcohol spectrum disorders: Characterizing caregivers' use of PBS using follow-up clinical trial data.

BACKGROUND: Fetal alcohol spectrum disorders (FASD) are associated with neurodevelopmental challenges leading to difficulties with everyday life tasks. The Families Moving Forward (FMF) Program teaches caregivers to use positive behavior support (PBS), integrated with other techniques. However, it is unknown how caregivers retain and use these PBS strategies after the intervention. METHODS: About 4.5 months after completing the FMF Program, twenty-three caregivers of children with FASD aged 4-12 were interviewed about problem behaviors targeted during the FMF Program and their continued use of PBS strategies. Interviews were recorded and coded thematically by a five-coder team. Higher-level pattern codes were developed to facilitate themes across descriptive codes. RESULTS: Caregivers commonly targeted task incompletion and rule breaking, and problem behaviors were often complex or combined. Caregivers identified environmental and interpersonal triggers for problem behavior. They used many accommodations to prevent problem behaviors, most often related to task or environment simplification. Caregivers also used consequence-based strategies. CONCLUSIONS: This study is the first to characterize caregivers' use of PBS strategies for children with FASD using mixed methods. Problem behaviors such as rule breaking were more difficult to target. Caregivers found most success when using a combination of multiple different accommodations per problem behavior. WHAT THIS PAPER ADDS: This is the first study to use mixed methods to characterize how caregivers of children with fetal alcohol spectrum disorders (FASD) use positive behavior support (PBS) strategies to target problem behavior after completion of the empirically validated Families Moving Forward (FMF) Program. Among other techniques involved in the FMF Program, PBS strategies are taught to caregivers and are used to target two distinct, caregiver-identified problem behaviors. This data provides essential information about behaviors responsive to PBS supports, for children with FASD, to inform clinical intervention and research. Notably, multiple problem behaviors often occurred together, emphasizing complexity of behavior challenges in this population and the resulting need for individualized supports. This study is the first to describe commonly observed triggers (antecedents) and commonly used supports (accommodations) from the perspective of caregivers of children with FASD. Importantly, results indicate that use of a wide variety of accommodations, or antecedent-based strategies, are effective in supporting behavior in children with FASD. However, success was most common when caregivers used multiple accommodations for any given concerning behavior. Findings represent 'real-world' strategies caregivers use to support adaptive behavior in their children several months after completion of the FMF Program, suggesting these strategies are applicable to clinical practice.

Children and adolescents with fetal alcohol syndrome (FAS): better social and emotional integration after early diagnosis.

BACKGROUND: Alcoholism during pregnancy is one of the most common factors in western societies causing persisting congenital and multiple physical as well as neurological impairments. Finding the diagnosis at first sight puts medical professionals into a demanding situation. Therefore the objective of this study was to detect patients' developmental characteristics with the main focus on the necessity of the diagnosis itself. PATIENTS: 125 young patients, whose diagnosis of fetal alcohol syndrome (FAS) was made at the Muenster University Hospital, were followed up. METHODS: Biographic details such as living conditions, health, developmental problems and educational career were gathered using a structured interview. The diagnosis itself and the impact of this on the patients were also explored. RESULTS: Patients displayed characteristics of a less mature trait of character. The majority were looked after by foster parents. High rates of social and developmental problems could be found. The diagnosis was identified as a protective factor, with significantly better outcomes for patients being diagnosed in early childhood. A diagnosis established later in life was particularly helpful for the families and caregivers. Feelings of failure and self-blame could be diminished. CONCLUSIONS: The early detection of affected children has to be improved as receiving the correct diagnosis, despite the persistent impairments, is of major benefit for both patients and their families.

Young adults with Fetal Alcohol Syndrome (FAS): social, emotional and occupational development.

BACKGROUND: Maternal alcohol abuse during pregnancy causes physical, cognitive and behavioural impairments in the child. Deficits are irreversible and persist into adulthood. It was the objective of this study to investigate the specific problems and challenges faced by young adults with Fetal Alcohol Syndrome (FAS). PATIENTS: We examined the biographical development and living situation of 60 adult patients with FAS. In their childhood, all the patients had been diagnosed with FAS in the Muenster University hospital. METHODS: Using a structured interview, we asked for the occupational career, health and social problems as well as the current living conditions of the patients. RESULTS: Most patients lived in dependent circumstances. The occupational development was characterized by disruption and failure, and severe social problems were common in patients. 3 in 4 patients were victims of physical and sexual abuse. CONCLUSIONS: Due to the variety of impairments caused by prenatal alcohol exposure and the persisting inability to live independently, adults with FAS need intense care, support and assistance.

The effectiveness of a community-based intervention for parents with FASD.

The purpose of this study was to evaluate the effectiveness of the Step by Step program in which mentors work with parents affected by Fetal Alcohol Spectrum Disorder (FASD) on a one-to-one basis. Mentors help clients identify and work towards meeting their needs and achieving their goals. Data from 24 closed client files was collected and analyzed and as predicted, the program was effective in helping clients reduce their needs and achieve their goals. The clients' reason for leaving the program as well as whether or not they had a formal FASD diagnosis had an impact on their success in the program. Data collected on additional mental health issues, experience of abuse and addictions helped to characterize the sample of clients and correlations were found between clients' experience of abuse and their past and/or present addictions issues. Limitations of this study as well as future implications were also discussed.

The best possible start: A qualitative study on the experiences of parents of young children with or at risk for fetal alcohol spectrum disorders.

BACKGROUND: The developmental outcomes and life course trajectories of young children with or at-risk for fetal alcohol spectrum disorders (FASD) can be optimized when individual and family needs are identified early and met with family-centered early intervention (EI) services. However, little is known about access to and quality of EI services with this high-needs population. METHOD: Twenty-five biological or adoptive parents of children with or at high risk for FASD, living in the greater area of Seattle, Washington participated in this qualitative study. Three focus groups were conducted using a semi-structured interview guide. Participants described their experience with EI, as well as other supports and challenges faced in their child's first three years of life. Interviews were audio recorded, transcribed verbatim and coded using phenomenological methods. Themes that were consistent across participant groups emerged from the data, as well as themes that showed differences among participant experiences. RESULTS: Common EI supports and needs between biological and adoptive parent groups were identified. In addition, perspectives and needs unique to each parent group were revealed. Themes were identified and organized into three categories: (1) child needs; (2) parent needs and priorities; and (3) EI capacity. When parents talked about their child's cognitive, physical, communication or adaptive development, they all discussed how EI was meeting those needs. In contrast, when parents expressed concern for their child's social-emotional development, a description of how EI was supporting these needs was missing from the conversation. Parents appreciated when EI providers were truthful, provided anticipatory guidance, and connected them with supports for their own social-emotional well-being. Yet there were unmet needs for respite care, and parents expressed that support for basic needs related to child or family survival was not consistently recognized as a top priority for families. This high-risk group of young children and their parents also encountered a multitude of transitions in their child's early years and later. Parents wanted more support navigating these transitions as they entered or moved through different systems of care. CONCLUSIONS: Parents appreciated and endorsed the importance of EI with its provision of individualized, family-centered supports and resources. Examination of the gaps and unmet needs that are common and distinct underscore the importance of an FASD-informed approach to EI. Study findings provide insight into areas for which EI enhancements could be developed in order to tailor supports for the complex needs of this diverse population of children and parents.

Systematic review of interventions for children with Fetal Alcohol Spectrum Disorders.

BACKGROUND: Children with Fetal Alcohol Spectrum Disorders (FASD) may have significant neurobehavioural problems persisting into adulthood. Early diagnosis may decrease the risk of adverse life outcomes. However, little is known about effective interventions for children with FASD. Our aim is to conduct a systematic review of the literature to identify and evaluate the evidence for pharmacological and non-pharmacological interventions for children with FASD. METHODS: We did an electronic search of the Cochrane Library, MEDLINE, EMBASE, PsychINFO, CINAHL and ERIC for clinical studies (Randomized controlled trials (RCT), quasi RCT, controlled trials and pre- and post-intervention studies) which evaluated pharmacological, behavioural, speech therapy, occupational therapy, physiotherapy, psychosocial and educational interventions and early intervention programs. Participants were aged under 18 years with a diagnosis of a FASD. Selection of studies for inclusion and assessment of study quality was undertaken independently by two reviewers. Meta-analysis was not possible due to diversity in the interventions and outcome measures. RESULTS: Twelve studies met the inclusion criteria. Methodological weaknesses were common, including small sample sizes; inadequate study design and short term follow up. Pharmacological interventions, evaluated in two studies (both RCT) showed some benefit from stimulant medications. Educational and learning strategies (three RCT) were evaluated in seven studies. There was some evidence to suggest that virtual reality training, cognitive control therapy, language and literacy therapy, mathematics intervention and rehearsal training for memory may be beneficial strategies. Three studies evaluating social communication and behavioural strategies (two RCT) suggested that social skills training may improve social skills and behaviour at home and Attention Process Training may improve attention. CONCLUSION: There is limited good quality evidence for specific interventions for managing FASD, however seven randomized controlled trials that address specific functional deficits of children with FASD are underway or recently completed.

Caregiver and family quality of life for children with fetal alcohol spectrum disorder.

BACKGROUND: Fetal alcohol spectrum disorder (FASD) is a common neurodevelopmental condition. Given that individuals with FASD can experience lifelong challenges, one field of research that could be applicable is the paediatric chronic health literature. AIMS: The aim of the current study was to investigate the utility of the Pediatric Quality of Life Inventory (PedsQL) Family Impact Module, designed to measure the impact of paediatric chronic health conditions on caregivers and families. METHODS AND PROCEDURES: 109 caregivers of children with FASD completed an online survey that assessed a range of areas including, caregiver and family quality of life, caregiver mental health and child behaviour. OUTCOMES AND RESULTS: Overall, caregivers reported the areas most impacted on the PedsQL module were Family Daily Activities and Worry. Caregiver's country of residence, mental health, child gender, and level of child behaviour problems were found to be predictors of caregiver and family quality of life. CONCLUSIONS AND IMPLICATIONS: The results demonstrate that there are multidimensional challenges for caregivers and families. These findings have important implications for policy and practice regarding the provision of supports and services for children with FASD and their families.

Two case reports of fetal alcohol syndrome: broadening into the spectrum of cardiac disease to personalize and to improve clinical assessment.

BACKGROUND: Fetal alcohol spectrum disorder (FASD) refers to a broad spectrum of disabilities, in infants and children, resulting from moderate to excessive prenatal alcohol exposure. Significant associations with alcohol exposure were already reported with congenital structural heart defects: i.e. ventricular septal defects, atrial septal defects, conotruncal defects. CASES PRESENTATION: We describe two cases of children with FASD, both admitted to the Center for Rare Diseases and Birth Defects of Policlinico Universitario Agostino Gemelli, in whom asymptomatic cardiac rhythm alterations were detected in absence of structural cardiovascular system anomalies or cardiac channelopathies. CONCLUSIONS: No other reports about cardiac rhythm anomalies in individuals affected by FASD are actually available from the literature. We would like to make an alert for clinician, given the possibility of finding anomalies of heart conduction and rhythm in children affected by FASD even without structural congenital heart disease.

The effect of rehearsal training on working memory span of children with fetal alcohol spectrum disorder.

A key area of weakness in individuals with fetal alcohol spectrum disorder (FASD) is working memory, thus the goal of this study was to determine whether teaching children (aged 4-11) with FASD verbal rehearsal would increase their memory. Rehearsal training has been effective in other populations with working memory difficulties, so we hypothesized that children with FASD would also benefit from rehearsal training. Children were divided into an Experimental group, who received rehearsal training and a Control group, who did not receive training. All children were tested on digit span tasks over three sessions: a pretest (baseline) and then post-test 1 and post-test 2 (where only the Experimental group received rehearsal training). The Experimental group showed a significant increase in performance across session but the Control group did not. Children in the Experimental group performed significantly higher than the Control group on post-test 2 but not on the pretest or post-test 1. More children in the Experimental group showed behavioral evidence and self-report of rehearsal after training. Rehearsal training was successful at increasing the memory for numbers among children with FASD and may help to ameliorate working memory difficulties in FASD, ultimately supporting academic and developmental growth of children with FASD.

GoFAR: improving attention, behavior and adaptive functioning in children with fetal alcohol spectrum disorders: Brief report.

OBJECTIVE: This brief report describes the GoFAR intervention designed to improve attention, behavior, and adaptive functioning in children with FASD, ages 5 to 10 years. METHODS: Thirty children were randomized to one of three conditions: GoFAR; FACELAND, and CONTROL; 25 completed the interventions. Over 10 sessions children and caregivers learned a metacognitive strategy (FAR) designed to improve cognitive control of behavior and adaptive functioning and practiced it during behavior analog therapy. Attention, behavior problems, and adaptive skills were measured pre- and post-intervention. RESULTS: From pre- to post-testing the GoFAR intervention group improved on the Test of Variables of Attention (TOVA). Both intervention groups improved in Daily Living Skills. CONCLUSION: This pilot study demonstrated that children with FASD and their caregivers benefit from a focused intervention designed to improve effortful control of behavior. The study suggests the need for a larger clinical trial to evaluate the intervention's effectiveness.

Investigating a theatre-based intervention for Indigenous youth with fetal alcohol spectrum disorder: Exploration d'une intervention basée sur le théâtre auprès de jeunes Autochtones atteints du syndrome d'alcoolisme fœtal.

BACKGROUND: Theatre-based interventions use artistic media to facilitate social and emotional awareness and have therapeutic benefits for persons with developmental disabilities and mental health problems. The role of these interventions with Indigenous youth who have emotional, behavioural, and cognitive sequelae related to fetal alcohol spectrum disorder (FASD) has not been explored. PURPOSE: The purpose of this study was to explore the experiences and acceptability of a theatre-based approach for facilitating social communication and engagement in youth with FASD. METHOD: Participants were three Indigenous youth with FASD. A qualitative exploration of the experiences and acceptability of the intervention was conducted via focus groups held 2 weeks post-program participation with the participants, their caregivers, and program facilitators. The transcripts were analyzed using an inductive thematic approach. FINDINGS: Our results identified perceived postintervention improvements in participants' development of self-esteem, social skills, and emotional awareness. IMPLICATIONS: A theatre-based arts intervention has the potential to support improvements in social skills for youth with FASD.

Mathematics intervention for children with fetal alcohol spectrum disorder: A replication and extension of the math interactive learning experience (MILE) program.

BACKGROUND: Individuals with fetal alcohol spectrum disorders (FASD) experience deficits in behavior, cognition, and academic functioning resulting from prenatal alcohol exposure (PAE). Although receiving intervention for developmental disabilities is a strong protective factor against negative outcomes in FASD, intervention research in this population is in its infancy. AIMS: The purpose of this study was to replicate and extend a mathematics intervention, the Math Interactive Learning Experience (MILE) program, which was developed in the USA specifically for children with FASD. METHODS: Twenty-eight Canadian children aged 4-10 years with confirmed PAE or an FASD diagnosis were assigned to either the MILE intervention or a contrast intervention. RESULTS: Following a relatively brief, individualized, one-on-one intervention, children in the MILE group demonstrated significantly greater changes in math achievement compared to the contrast group. Significant changes in other cognitive functions were not observed. Older age, lower IQ, and confirmed PAE but no FASD diagnosis were associated with greater math achievement change in the MILE group. CONCLUSIONS: The replication and extension of the math intervention appears to have significant, positive impact on mathematics achievement scores of children with PAE and FASD.

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

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

FASD: what types of intervention and rehabilitation are useful?

Fetal alcohol spectrum disorders (FASD) occurs worldwide when children are prenatally exposed to alcohol. This paper discusses recent findings regarding the neuropsychological and behavioral effects of prenatal alcohol exposure and how it impacts the developmental and functional abilities of children with FASD. Specifically, recent research focus has concentrated on studies to elucidate a neurobehavioral phenotype for the alcohol-exposed population. As a result, the FASD field has learned what types of neurobehavioral issues occur most frequently with these children. This paper discusses how that information can be used to inform school assessment, intervention planning, and support. Strategies for functional assessment, individualized planning, structured teaching, and developments in cognitive-behavioral methods are described.