Health, Social, Education, and Justice Outcomes of Manitoba First Nations Children Diagnosed with Fetal Alcohol Spectrum Disorder: A Population-Based Cohort Study of Linked Administrative Data.

OBJECTIVE: To examine health services, social services, education, and justice system outcomes among First Nations children and youth with fetal alcohol spectrum disorder (FASD). METHODS: In this retrospective cohort study, health and social services, education, and justice data were linked with clinical records on First Nations (FN) individuals aged 1 to 25 and diagnosed with FASD between 1999 and 2010 (n = 743). We compared the FN FASD group to non-FN individuals with FASD (non-FN FASD; n = 315) and to First Nations individuals (matched on age, sex, and income) not diagnosed with FASD (FN non-FASD; n = 2229). Rates and relative risks (RRs) were calculated using generalized linear models. RESULTS: FN FASD individuals had similar health services use to non-FN FASD individuals but had greater involvement with child welfare (RR, 1.20; 95% confidence interval [CI], 1.02 to 1.41) and the justice system (RR, 1.37; 95% CI, 1.07 to 1.74) and were more likely to be charged with a crime (RR, 1.40; 95% CI, 1.05 to 1.86). There were no suicides/suicide attempts among the non-FN FASD individuals during the study, but the crude rate/100 person-years of suicides among FN FASD individuals (0.22 for females; 1.06 for males) was substantially higher than for FN non-FASD individuals (0.08 for females; 0.32 for males). There were no significant differences between groups in the education outcomes we measured. CONCLUSIONS: Young people with FASD are at risk for poor health, education, and social outcomes, but First Nations young people with FASD face comparably higher risks, particularly with child welfare and justice system involvement. The study emphasizes a critical need for appropriate resources for First Nations children with FASD.

The Psychiatric Morbidity of Women Who Give Birth to Children with Fetal Alcohol Spectrum Disorder (FASD): Results of the Manitoba Mothers and FASD Study.

OBJECTIVE: To investigate differences in physician-diagnosed psychiatric disorders between women who gave birth to children with a fetal alcohol spectrum disorder (FASD) diagnosis (study group) compared to women who gave birth to children without FASD (comparison group). METHODS: We linked population-level health and social services data to clinical data on FASD diagnoses to identify study group ( n = 702) and comparison group ( n = 2097) women matched 1:3 on date of birth of index child, region of residence, and socioeconomic status. Regression modeling produced relative rates (RRs) for outcomes. RESULTS: Mothers who gave birth to children with FASD had higher adjusted rates of substance use disorder (RR, 12.65; 95% confidence interval [CI], 8.99-17.80), personality disorder (RR, 12.93; 95% CI, 4.88-34.22), and mood and anxiety disorders (RR, 1.75; 95% CI, 1.49-2.07) before the pregnancy of the child. These mothers also had higher adjusted rates of maternal psychological distress during pregnancy (RR, 5.35; 95% CI, 4.58-6.35) and higher rates of postpartum psychological distress (RR, 1.71; 95% CI, 1.53-1.90). These women also had higher adjusted rates for antidepressant prescriptions before, during, and after the pregnancy. CONCLUSIONS: A significant psychiatric burden exists for women giving birth to children with FASD. Clinicians should recognise the high rates of psychiatric concerns facing mothers who give birth to children with FASD and should offer treatment and support to these women to improve their health and well-being and prevent further alcohol-exposed pregnancies.

Integrating care for individuals with FASD: results from a multi-stakeholder symposium.

BACKGROUND: Fetal Alcohol Spectrum Disorder (FASD) has a significant impact on communities and systems such as health, education, justice and social services. FASD is a complex neurodevelopmental disorder that results in permanent disabilities and associated service needs that change across affected individuals' lifespans. There is a degree of interdependency among medical and non-medical providers across these systems that do not frequently meet or plan a coordinated continuum of care. Improving overall care integration will increase provider-specific and system capacity, satisfaction, quality of life and outcomes. METHODS: We conducted a consensus generating symposium comprised of 60 experts from different stakeholder groups: Allied & Mental Health, Education, First Nations & Métis Health, Advocates, Primary Care, Government Health Policy, Regional FASD Coordinators, Social Services, and Youth Justice. Research questions addressed barriers and solutions to integration across systems and group-specific and system-wide research priorities. Solutions and consensus on prioritized lists were generated by combining the Electronic Meeting System approach with a modified 'Nominal Group Technique'. RESULTS: FASD capacity (e.g., training, education, awareness) needs to be increased in both medical and non-medical providers. Outcomes and integration will be improved by implementing: multidisciplinary primary care group practice models, FASD system navigators/advocates, and patient centred medical homes. Electronic medical records that are accessible to multiple medical and non-medical providers are a key tool to enhancing integration and quality. Eligibility criteria for services are a main barrier to integration across systems. There is a need for culturally and community-specific approaches for First Nations communities. CONCLUSIONS: There is a need to better integrate care for individuals and families living with FASD. Primary Care is well positioned to play a central and important role in facilitating and supporting increased integration. Research is needed to better address best practices (e.g., interventions, supports and programs) and long-term individual and family outcomes following a diagnosis of FASD.

"Giving voice": Evaluation of an integrated telehealth community care model by parents/guardians of children diagnosed with fetal alcohol spectrum disorder in Manitoba.

BACKGROUND: In 2000, Manitoba began utilizing telehealth services for the assessment, diagnosis and follow-up of fetal alcohol spectrum disorders (FASDs). Since that time, the use of telehealth in Manitoba has expanded to the delivery of education and support to families caring for children with FASD in rural and remote areas of the province. The purpose of this study was to expand on a previous evaluation through a focus on the participant experience. Our objectives were thus to explore the experience of families with the telehealth process and to examine the use of telehealth in diagnostic assessment as well as follow-up post-clinical assessment. MATERIALS AND METHODS: Sixteen semistructured interviews were conducted with families who had participated in at least one diagnostic assessment and/or individual or group follow-up via telehealth offered through the Manitoba FASD Centre. RESULTS: The majority of participants reported being happy with their experience(s) using telehealth for assessment, diagnosis, and/or follow-up support. Two general themes emerged from the data. The first theme is focused on the value of telehealth use for families with children living with FASD, whereas the second theme presents various needs of this client group. CONCLUSIONS: This study provides support for the use of telehealth as an effective technology beyond diagnosis for individuals with FASD and their families. Families support the utilization of this technology and, despite its minor shortcomings, appreciate the flexibility of telehealth, which allows them to remain in their home communities, connected to their families and support systems.

Using telehealth for assessment of fetal alcohol spectrum disorder: the experience of two Canadian rural and remote communities.

INTRODUCTION: Telehealth has been used for fetal alcohol spectrum disorder (FASD) diagnostic assessment in select Manitoban communities since 2000. OBJECTIVE: The purpose of this study was to evaluate the FASD telehealth program within two rural and remote Northern Manitoban communities by comparing community practices from the perspective of professionals working with the FASD diagnostic clinics in these communities. Recommendations for the further development of FASD assessment by telehealth were made to further improve current implementation and guide expansion of the FASD telehealth program within the province. METHODOLOGY: Semistructured interviews were conducted from October 19 to December 11, 2009. Participants (N = 26) were comprised of professionals, including those in the education, social services, and health sectors. RESULTS AND RECOMMENDATIONS: Two themes emerged from the data and covered the perceived strengths and drawbacks with the program, and meaningful suggestions to improve the service. Participants regarded the FASD telehealth program as successful and useful, especially given the remote location of the communities and the lack of on-site services. Recommendations addressing the barriers pertaining to the process were made from the study's findings and available scientific literature. CONCLUSIONS: This study will provide a solid basis for the successful further development of the FASD telehealth programs.

Prenatal care of women who give birth to children with fetal alcohol spectrum disorder in a universal health care system: a case-control study using linked administrative data.

BACKGROUND: Few studies have investigated prenatal care use among women who use alcohol during pregnancy. The objective of this study was to investigate rates of prenatal care usage of women who have given birth to children with fetal alcohol spectrum disorder (FASD). METHODS: We conducted a case-control study of women with children born in Manitoba between Apr. 1, 1984, and Mar. 31, 2012, with follow-up until 2013, using linkable administrative data. The study group included women whose child(ren) was (were) diagnosed with FASD (n = 702) between Apr. 1, 1999, and Mar. 31, 2012, at a centralized diagnostic clinic. The comparison group included women whose child(ren) did not have an FASD diagnosis (n = 2097), exact matched on the index child's birthdate, postal code and socioeconomic status. Adequacy of prenatal care was defined using the Revised Graduated Prenatal Care Utilization Index. RESULTS: Women in the study group had lower socioeconomic status than women in the comparison group and were more likely to have mental disorders and involvement with the child welfare system. Rates of inadequate prenatal care were higher among women in the study group (adjusted relative risk 2.47, 95% confidence interval [CI] 2.08-2.94), as were rates of no prenatal care (adjusted relative risk 3.55, 95% CI 2.42-5.22). In the study group, 41% of women accessed inadequate or no prenatal care, and 59% received intermediate, adequate or intensive prenatal care. INTERPRETATION: Women who give birth to children with FASD have higher rates of inadequate prenatal care and significant social complexities. Socioeconomic disparities in the use of prenatal care should be addressed; multisector interventions are needed that facilitate the uptake of prenatal care by high-risk women who use alcohol.

Suicide and suicide attempts among women in the Manitoba Mothers and Fetal Alcohol Spectrum Disorder cohort: a retrospective matched analysis using linked administrative data.

BACKGROUND: Women who give birth to children with fetal alcohol spectrum disorder (FASD) may be at increased risk for suicide; however, there are few data in this area. The objective of this study was to compare rates of suicide between women who had given birth to children with FASD and women who had not given birth to children with FASD during critical periods in their lives, including before pregnancy, during pregnancy, during the postpartum period (maternal death) and until the end of the study period. METHODS: We conducted a retrospective cohort analysis of women with children born in Manitoba between Apr. 1, 1984, and Mar. 31, 2012 in whom FASD was diagnosed between Apr. 1, 1999, and Mar. 31, 2012, with follow-up until Dec. 1, 2013 (FASD group; n = 702). We generated a comparison group of women who had not given birth to children with FASD (n = 2097), matched up to 1:3 on date of birth of the index child, socioeconomic status and region of residence. We used linked administrative data to investigate suicide attempt and completion rates in the 2 groups. Regression modelling produced relative rates (RRs) adjusted for socioeconomic status and age at birth of the index child and was used to assess suicide risk. RESULTS: The 2799 participants produced 40 390.21 person-years until the end of the study period. Compared to the comparison group, the FASD group had higher rates of suicide completion (adjusted RR 6.20 [95% confidence interval (CI) 2.36-16.31]), a higher number of women who attempted suicide after the postpartum period until the end of the study period (adjusted RR 4.62 [95% CI 2.53-8.43]) and a higher number of attempts after the postpartum period until the end of the study period (adjusted RR 3.92 [95% CI 2.30-6.09]). INTERPRETATION: This study identified a group of women with increased rates of social complexities, mental disorders and alcohol use, which places them at risk for suicide. Interventions are needed that screen for suicidal behaviour in women who are at high risk to consume alcohol during pregnancy and have mental disorders.

A descriptive Study of a Community-Based Home-Visiting Program with Preschool Children Prenatally Exposed to Alcohol.

BACKGROUND: Research suggests that prenatal alcohol exposure (PAE) and Fetal Alcohol Spectrum Disorder (FASD) increases the risk of insecure caregiver-child attachment and related negative child emotional-behavioural outcomes. Research also shows positive effects of attachment-focused interventions in preventing disrupted caregiver-child attachment relationships; however, such interventions have not been specifically adapted for children with FASD. OBJECTIVES: This paper describes the implementation, challenges, and results of a community home-based attachment intervention, Circle of Security® (COS), with preschool children affected by PAE/FASD in Manitoba, Canada. METHODS: Twelve caregiver-child dyads completed the FASD adapted COS intervention. Children's ages ranged from 2-5 years. RESULTS: Results support a positive influence of this individualized intervention on child behaviour and parent efficacy. There was a significant reduction in parent stress that was maintained at 3-month follow-up, and a clinically significant reduction in child behavioural issues was noted. Children showed increased ability to communicate their needs more effectively to their parent. Parents also showed an improvement in their ability to attend to their child's cues. CONCLUSIONS: This study supports the use of community home-based attachment intervention for caregivers of children with PAE/FASD.

Manitoba mothers and fetal alcohol spectrum disorders study (MBMomsFASD): protocol for a population-based cohort study using linked administrative data.

INTRODUCTION: Fetal alcohol spectrum disorder (FASD) is a significant public health concern. To prevent FASD, factors that place women at risk for giving birth to children with FASD must be investigated; however, there are little data in this area. This paper describes the development of the Manitoba mothers and FASD study, a retrospective cohort of mothers whose children were diagnosed with FASD, generated to investigate: (1) risk factors associated with giving birth to children with FASD; (2) maternal physical and health outcomes, as well as the usage of health and social services. METHODS: The study population will be identified by linking children diagnosed with FASD from a provincially centralised FASD assessment clinic (from 31 March 1999 to 31 March 2012) to their birth mothers using de-identified administrative health data housed at the Manitoba Centre for Health Policy. Preliminary analysis has identified over 700 mothers, which is the largest sample size in this field to date. A comparison cohort of women with children who did not have an FASD diagnosis matched on the region of residence, date of birth of child with FASD and socioeconomic status will be generated to compare exposures and outcomes. Potential demographic, socioeconomic, family history, and physical and mental health risk factors will be investigated by linking a range of health and social databases, furthering insight into the root causes of drinking during pregnancy. The longitudinal data will allow us to document the usage patterns of healthcare and social services throughout significant periods in these women's lives to identify opportunities for prevention. ETHICS AND DISSEMINATION: Ethical approval has been obtained by the University of Manitoba's Health Research Ethics Board and the Manitoba Health Information Privacy Committee. Dissemination of study results will include engagement of stakeholders and policymakers through presentations and reports for policymakers, in parallel with scientific papers.

Visual search for feature conjunctions: an fMRI study comparing alcohol-related neurodevelopmental disorder (ARND) to ADHD.

BACKGROUND: Alcohol-related neurodevelopmental disorder (ARND) falls under the umbrella of fetal alcohol spectrum disorder (FASD). Diagnosis of ARND is difficult because individuals do not demonstrate the characteristic facial features associated with fetal alcohol syndrome (FAS). While attentional problems in ARND are similar to those found in attention-deficit/hyperactivity disorder (ADHD), the underlying impairment in attention pathways may be different. METHODS: Functional magnetic resonance imaging (fMRI) and diffusion tensor imaging (DTI) was conducted at 3 T. Sixty-three children aged 10 to 14 years diagnosed with ARND, ADHD, and typically developing (TD) controls performed a single-feature and a feature-conjunction visual search task. RESULTS: Dorsal and ventral attention pathways were activated during both attention tasks in all groups. Significantly greater activation was observed in ARND subjects during a single-feature search as compared to TD and ADHD groups, suggesting ARND subjects require greater neural recruitment to perform this simple task. ARND subjects appear unable to effectively use the very efficient automatic perceptual 'pop-out' mechanism employed by TD and ADHD groups during presentation of the disjunction array. By comparison, activation was lower in ARND compared to TD and ADHD subjects during the more difficult conjunction search task as compared to the single-feature search. Analysis of DTI data using tract-based spatial statistics (TBSS) showed areas of significantly lower fractional anisotropy (FA) and higher mean diffusivity (MD) in the right inferior longitudinal fasciculus (ILF) in ARND compared to TD subjects. Damage to the white matter of the ILF may compromise the ventral attention pathway and may require subjects to use the dorsal attention pathway, which is associated with effortful top-down processing, for tasks that should be automatic. Decreased functional activity in the right temporoparietal junction (TPJ) of ARND subjects may be due to a reduction in the white matter tract's ability to efficiently convey information critical to performance of the attention tasks. CONCLUSIONS: Limited activation patterns in ARND suggest problems in information processing along the ventral frontoparietal attention pathway. Poor integrity of the ILF, which connects the functional components of the ventral attention network, in ARND subjects may contribute to the attention deficits characteristic of the disorder.

Smith-Lemli-Opitz syndrome: new mutation with a mild phenotype.

Smith-Lemli-Opitz syndrome (SLOS) (Online Mendelian Inheritance in Man, OMIM, 2001, http://www.ncbi.nlm.nih.gov/omim/ for SLOS, MIM 270400) is an autosomal recessive disorder of cholesterol biosynthesis caused by mutations of the 3beta-hydroxysterol Delta(7)-reductase gene, DHCR7. We report on a female infant with an exceptionally mild phenotype of SLOS, in whom molecular studies identified a new mutation in DHCR7. The proposita initially presented with feeding difficulties, failure to thrive, hypotonia, mild developmental delay, and oral tactile aversion. She had minor facial anomalies and 2-3 syndactyly of her toes in both feet. The plasma cholesterol was borderline low at 2.88 mmol/L (normal 2.97-4.40 mmol/L). Elevated plasma 7-dehydrocholesterol level of 200.0 micromol/L confirmed the clinical diagnosis of SLOS. Molecular analysis demonstrated compound heterozygosity for IVS8-1G -->C and Y280C, a new missense mutation in DHCR7. Since the other mutation in this patient is a known null mutation, this newly discovered mutation is presumably associated with significant residual enzyme activity and milder expression of clinical phenotype.

Use of health, education, and social services by individuals with fetal alcohol spectrum disorder.

BACKGROUND: Fetal Alcohol Spectrum Disorder (FASD) is the leading cause of intellectual disability in western society, presenting a significant burden on health, education and social services. Quantifying the burden of FASD is important for service planning and policy and program development. OBJECTIVE: To describe the health, education and social service use of individuals with FASD to provide an indication of the burden of service use of the disorder. METHODS: Using a matched-cohort design health, education and social service data were linked with clinical records on individuals 6+ years diagnosed with FASD between 1999/2000-2009/10 (N=717). Matching was 2:1 with a general population (gPop) and asthma group by age, sex and area-level income. Adjusted rates and relative risks were calculated using Generalized Linear Models. RESULTS: Hospitalizations were higher in the FASD compared to gPop (adjusted relative risk=3.44 (95% confidence interval=2.29, 5.17)) and asthma (2.87 (1.94, 4.25)) groups, whereas for physician visits and overall prescriptions, the FASD group differed from only the gPop group (1.58 (1.34, 1.84); 1.44 (1.22, 1.72), respectively). Antibiotics, pain killers and anti-psychotics were similar across groups whereas antidepressants and psychostimulants were higher in the FASD group (antidepressants: FASD vs. gPop 8.76 (2.82, 27.21); FASD vs. asthma 2.10 (1.15, 3.83); psychostimulants: FASD vs. gPop 5.78 (2.89, 11.57); FASD vs. asthma 2.47 (1.37, 4.47)). Attention-deficit\hyperactivity disorder was higher in the FASD than the gPop and asthma groups (6.41 (3.29, 12.49); 3.12 (1.97, 4.93), respectively). Education and social service use was higher for the FASD than either of the other groups for all measures (FASD vs. gPop and FASD vs. asthma, respectively for: grade repetition 3.06 (1.58, 5.94); 3.48 (1.79, 6.78); receipt of any special education funding 9.22 (6.23, 13.64); 6.10 (4.14, 8.99); family receipt of income assistance 1.74 (1.33, 2.27); 1.89 (1.45, 2.47); child in care 13.19 (5.84, 29.78);10.70 (4.80, 23.88); and receipt of child welfare services 5.70 (4.21, 7.71); 4.94 (3.67, 6.66)). CONCLUSION: The health, education and social service utilization burden of individuals with FASD is substantial, greater than that of individuals in the general population and with chronic illness (i.e., asthma). The findings highlight the need for multisystem supports for those with FASD, and comprehensive prevention programs.

Comparison of spatial working memory in children with prenatal alcohol exposure and those diagnosed with ADHD; A functional magnetic resonance imaging study.

BACKGROUND: Alcohol related neurodevelopmental disorder (ARND) falls under the umbrella of fetal alcohol spectrum disorder (FASD), but individuals do not demonstrate the facial characteristics associated with fetal alcohol syndrome (FAS), making diagnosis difficult. While attentional problems in ARND are similar to those found in attention-deficit/hyperactivity disorder (ADHD), the underlying impairment in attention pathways may be different. METHODS: Functional magnetic resonance imaging (fMRI) of a working memory (1-back) task of 63 children, 10 to 14 years old, diagnosed with ARND and ADHD, as well as typically developing (TD) controls, was conducted at 3 T. Diffusion tensor imaging (DTI) data were also acquired. RESULTS: Activations were observed in posterior parietal and occipital regions in the TD group and in dorsolateral prefrontal and posterior parietal regions in the ARND group, whereas the ADHD group activated only dorsolateral prefrontal regions, during the working memory component of the task (1-back minus 0-back contrast). The increases in frontal and parietal activity were significantly greater in the ARND group compared to the other groups. This increased activity was associated with reduced accuracy and increased response time variability, suggesting that ARND subjects exert greater effort to manage short-term memory load. Significantly greater intra-subject variability, demonstrated by fMRI region-of-interest analysis, in the ADHD and ARND groups compared to the TD group suggests that moment-to-moment lapses in attention contributed to their poorer task performance. Differences in functional activity in ARND subjects with and without a diagnosis of ADHD resulted primarily from reduced activation by the ARND/ADHD + group during the 0-back task. In contrast, children with ADHD alone clearly showed reduced activations during the 1-back task. DTI analysis revealed that the TD group had significantly higher total tract volume and number of fibers than the ARND group. These measures were negatively correlated with errors on the 1-back task, suggesting a link between white matter integrity and task performance. CONCLUSIONS: fMRI activations suggest that the similar behavior of children with ARND and ADHD on a spatial working memory task is the result of different cognitive events. The nature of ADHD in children with ARND appears to differ from that of children with ADHD alone.

Evaluation of spatial working memory function in children and adults with fetal alcohol spectrum disorders: a functional magnetic resonance imaging study.

Magnetic resonance imaging (MRI) and functional MRI studies involving n-back spatial working memory (WM) tasks were conducted in adults and children with Fetal Alcohol Spectrum Disorders (FASD), and in age- and sex-matched controls. FMRI experiments demonstrated consistent activations in regions of the brain associated with working memory. Children with FASD displayed greater inferior-middle frontal lobe activity, while greater superior frontal and parietal lobe activity was observed in controls. Control children also showed an overall increase in frontal lobe activity with increasing task difficulty, while children with FASD showed decreased activity. FASD adults demonstrated less functional brain activity overall, but greater inferior-middle frontal lobe activity during the simpler tasks, relative to controls. Control adults demonstrated greater inferior frontal activity with increasing task difficulty, while this pattern was not consistently observed in FASD adults. All four groups showed increasing activity with increases in task difficulty in the parietal and frontal regions at more superior slice levels. The results suggest impairment in spatial working memory in those with FASD that does not improve with age, and that fMRI may be useful in evaluation of brain function in these individuals.