Increased Mortality Risk in Children with Fetal Alcohol Spectrum Disorders: A Scoping Review

Objective: Fetal Alcohol Spectrum Disorders (FASDs) are common, often undiagnosed, lifelong developmental disorders that result from prenatal alcohol exposure. FASD is present at birth and typically identified around seven years of age. The most severe outcome in cases of FASD is mortality. The purpose of this scoping review is to 1) use a systematic review to provide an estimated mortality proportion for children with FASD, and 2) update a study published in 2014 by reviewing published reports of mortality in individuals diagnosed with FASD. Method: A search of PubMed, CINAHL, and Google Scholar for reports published between 2013 and 2023 on mortality in individuals with FASD. Results: Three population-based studies have reported on all-cause mortality rates, finding a combined mortality rate of 10.9%, a 2.63 fold (95% CI: 2.61 to 2.65) increase in mortality risk over the general population. Since 2016, this review identified only eight new cases meeting the study inclusion criteria. The reported causes of death were five cases of pneumonia, and one case each of failure to thrive and dehydration, intestinal dilatation and asphyxiation caused by overeating due to pica, and acute gastric volvulus. Discussion: While current research suggests a diagnosis of FASD is associated with a 2.6-fold increase in mortality risk, this is likely an underestimation, as most cases of FASD-related mortality go unreported. Globally, about 1 new case is reported every 15 months. However, in the United States alone, between 1752 to 4400 FASD related deaths occur annually. Our review suggests that FASD is rarely identified as a causal or contributing factor in deaths of children and adolescents, resulting in a substantial undercount of FASD-related deaths. Increased attention to the role of FASD in infant and child mortality case reviews, child death review committee reports, and mortality reviews is needed.

Is a Mini-Screen for Fetal Alcohol Spectrum Disorder Feasible?

Background: Fetal Alcohol Spectrum Disorders (FASDs) are a global public health concern with lifelong consequences for affected individuals. Recent prevalence studies suggest FASD prevalence rates range from 1-5% among school age children. Most people with FASD are not correctly diagnosed and inadequate screening to identify patients with increased risk may contribute to under-diagnosis. This study developed a 10-item screening tool for FASD and examined its feasibility. Methods: The sample consisted of 355 children who had been evaluated at an FASD clinic. Data from the 33-item Alcohol Related Neurodevelopmental Disorder Behavioral Checklist was used to develop a brief FASD screen by comparing the changes in Cronbach’s alpha for different combinations of items. The validity of the brief scale was then further examined using receiving operating characteristic analyses. Results: The 10-item screen demonstrated acceptable sensitivity, specificity, and accuracy to identify children at high risk for FASD. The percentage correctly classified was 91.3 and the area under the receiving operating characteristic curve was 0.971. Conclusions: This feasibility study demonstrated that a screen for FASD consisting of 10 items with yes or no responses can be completed in 3 - 4 minutes. The tool is brief, with a low administration burden and has acceptable epidemiologic performance characteristics including accuracy. Future research should examine the performance of this tool when used in larger, community-based populations where screening for FASD would be appropriate.

Mortality in Fetal Alcohol Spectrum Disorders

Objective: Mortality in FASD has not been well studied. In this paper we review published reports of mortality in FASD. Method: We searched using Pub Med for all years in all languages for reports of all-cause mortality associated with any FASD. Results: We located 26 papers reporting on 57 deaths. Cause of death was reported for 49/57 cases (86%). The two most prevalent potential causes of death were malformations of the heart (37 of 49 cases, 75.5%) which varied from atrial septal defect or patent ductus arteriosus to tetralogy of Fallot, hypoplastic left heart, aortic arch interruption, etc. and brain malformations(25 of 49, 51%) including microcephaly, hydrocephalus, porencephaly, agenesis/absence of the corpus callosum and semilobar holoprosencephaly. In several cases potential causal findings overlapped. The three most frequent other causes of death were sepsis (7 cases, 14.3%), kidney malformations (7 cases, 14.3%), and cancer (4 cases, 8.2%). Over half the deaths (30/55, 54.5%) occurred in the first year of life. Discussion: We found that congenital heart disease was the most common cause of death in people with FASD. This may be due to an ascertainment bias since many of the published studies were focused on congenital heart disease in FASD. We conclude that FASD is frequently undetected in mortality events and could be a common finding in infant, child, adolescent and adult mortality.

Effectiveness of a Novel Low Cost Intervention to Reduce Prenatal Alcohol Exposure in the Congo

Objective: Determine the effectiveness of an intervention to reduce prenatal alcohol exposure in the Congo. Methods: We utilized a screening tool validated in the Congo to identify women who were drinking during pregnancy. The intervention was implemented by prenatal care providers comparing 162 women receiving the intervention with 58 (controls) who did not. The study endpoints were proportion of women who quit drinking, drinking days per week, drinks per drinking day, most drinks on any day, and number of binge episodes per week. Results: In the control group 36% of the women quit drinking compared to 54% in the intervention group (Chi-square 5.61;p = 0.02). The number of drinking days per week for the controls decreased by 50.1% compared to 68% for the intervention group (p = 0.008);drinks per drinking day for the controls decreased by 37% compared to 60.1% for the intervention group (p = 0.001);and most drinks on any occasion in the controls decreased by 38% compared to 61% for the intervention group (p = 0.004). Conclusions: This study demonstrates the effectiveness of a low cost in-office intervention to reduce prenatal alcohol exposure in the Congo. At $1.50 per beer, the reduction in drinks per week would more than pay for the cost of the intervention. In addition to efforts to reduce alcohol use prior to pregnancy in the Congo, providers can now offer an evidence based intervention to reduce exposure for women who continue to drink during pregnancy.

胎儿酒精谱系障碍的筛查工具与诊断标准

胎儿酒精谱系障碍是指母孕期酒精暴露导致的从胎儿轻度损伤到典型的胎儿酒精综合征的一系列障碍谱系,被公认为导致胎儿缺陷与儿童发育迟滞的主要原因之一,其症状包含四大类特征:胎儿宫内发育迟滞和/或出生后发育迟滞;中枢神经系统功能异常;颜面特征畸形(眼睑裂短、人中平以及上唇薄等);母孕期酒精暴露。本文介绍该胎儿酒精谱系障碍的筛查工具和诊断标准。

Pregnancy risk markers in Tourette syndrome: A systematic review

The published literature on the prevalence of pregnancy risk markers in patients with Tourette Syndrome (TS) was reviewed. PubMed was searched for papers describing studies of pregnancy risk markers in TS. All years and languages were searched, and the reference sections of each paper were also reviewed for additional citations. We identified 20 studies reporting on pregnancy risk markers in 1588 subjects with TS. Six studies used comparison populations and two utilized twins for comparisons. Three risk markers (decreased birth weight, father’s age, and number of prior terminations of pregnancy) were identified as possible risk markers for TS. To date, no pregnancy risk marker has been demonstrated to increase risk for development of TS, to increase syndromal severity, rates of comorbidity, or to increase duration of TS.

Relationships between Fetal Alcohol Spectrum Disorder, Adverse Childhood Experiences, and Neurodevelopmental Diagnoses

Objective: Children with fetal alcohol spectrum disorder (FASD) are overrepresented in early intervention programs, foster care, special education, juvenile corrections, and mental health services. In this study, we examine relationships between FASD and non-FASD controls for adverse childhood experiences (ACEs), and neurodevelopmental disorders. Methods: A chart review was conducted among patients seen at our clinic from 2010-2017 with data on FASD, ACEs, neurodevelopmental diagnoses, and foster or residential care placement available. Results: Relative risk for FASD was increased in patients with increased ACE scores (RR = 5.08), increased numbers of neurodevelopmental diagnoses (RR = 2.36), and patients who have been in foster or residential care (RR = 9.53). FASD risk increased as ACE scores or the number of neurodevelopmental diagnoses increased. Patients with any ACEs were 3.96 times more likely to have FASD, and those with eight or more ACEs were 6.31 times more likely to have FASD than those with no ACEs. Patients with three or more neurodevelopmental diagnoses were 6.55 times more likely to have FASD than those with two or fewer diagnoses. Nine or more diagnoses increased the risk for FASD ten-fold (RR = 10.91). Conversely, patients diagnosed with FASD were more likely to have at least three ACEs (RR = 3.71), at least five neurodevelopmental diagnoses (RR = 1.61), and high rates of previous foster or residential care placement (RR = 5.39). Conclusion: This study demonstrates that all children being considered for placement in foster care or residential should be screened for FASD.

Does Prenatal Exposure to Alcohol and Cigarette Smoking Increase Risk of Hospitalization for American Indian Infants

Background: Prenatal exposure to alcohol and tobacco are both associated with increased risk for adverse outcomes. American Indians experience disproportionate rates of adverse birth outcomes. The Indian people living in the Great Plains Area of the Indian Health Services have increased exposure to alcohol and smoking during pregnancy. This study aims to assess the relationship between prenatal alcohol and tobacco exposure and early hospitalization in a rural Tribal Nation. Methods: We used data from the prospective Safe Passage Study on pregnancies and hospitalizations within the first year of life. The relationship between time to hospitalization and prenatal tobacco/alcohol exposure, maternal demographics, post-traumatic stress disorder and depression were evaluated. The analysis utilized Chi-square tests. Kaplan-Meier curves were used to estimate the likelihood of hospitalization during the first year of life by substance use category. Results: From a sample of 432 infants, we identified 260 (60.2%) who had a hospitalization. We found no significant effect from that if mother drank, amount drank, amount smoked or if mother smoked, on the risk of hospitalization. We did identify an effect for exposure to alcohol or smoking and age to hospitalization. Exposure was associated with younger age at hospitalization. However, after controlling for mother’s age, this effect was not significant. Mother’s age was significant in all models of hospitalization risk from parental exposure. Discussion: Reducing prenatal exposure to tobacco and alcohol may decrease hospitalization rates for infants from this Tribal Nation, especially among young mothers.