Developmental, sensory and behavioral outcomes among infants and toddlers with prenatal alcohol exposure.

BACKGROUND: Prenatal alcohol exposure (PAE) can disrupt children's neurodevelopment and exert lasting influences on overall child well-being and family functioning. A comprehensive exploration of developmental outcomes in infants/toddlers with PAE seen for a diagnosis on the fetal alcohol spectrum can inform early identification and intervention. AIMS: To describe the prevalence and patterns of neurodevelopment, sensory processing, and emotional and behavioral functioning in a clinical sample of infants/toddlers with PAE. METHODS: In this retrospective analysis, clinical data from 125 infants/toddlers with PAE, aged 2-42 months, assessed at the University of Washington Fetal Alcohol Syndrome Diagnostic and Prevention Network clinic were analyzed. RESULTS: Seventy-four to 87% of infants/toddlers demonstrated delayed development in one or more domains of the Bayley Scales of Infant and Toddler Development (n = 125). Adverse developmental outcomes were significantly correlated with PAE and/or postnatal risk factors. All 93 infants/toddlers with a complete Infant/Toddler Sensory Profile obtained definite difference scores in at least one quadrant/section. Over half of infant/toddlers with a completed Child Behavior Checklist/1½- 5 had total problem scores in the borderline or clinical range. CONCLUSIONS: Findings suggest that several domains of child functioning may be vulnerable to the teratogenic impact of PAE, and that these delays are evident in the first years of life. Early screening, ongoing monitoring and comprehensive assessment is needed to facilitate earlier identification and guide clinical intervention.

Concerns and Strengths: Caregiver Perceptions of Their Infant/Toddler with Prenatal Alcohol Exposure.

Caregiver-reported assessments provide opportunities for caregivers to share concerns and identify the strengths of their infant/toddler regarding prenatal alcohol exposure (PAE). These insights may reveal under-recognized concerns and inform a strengths-based approach to early intervention. The purpose of this study was to describe the type and frequency of caregiver-reported concerns and strengths in a sample of infants/toddlers at the time of their fetal alcohol spectrum disorder (FASD) diagnostic evaluation. Caregivers' concerns and strengths were identified in the context of two parent-report questionnaires, the Infant Toddler Sensory Profile and Child Behavior Checklist/1½-5. By using content analysis, caregivers' open-ended responses were identified, coded, and analyzed. The frequencies of all the coded concerns and strengths were counted. The data were compared across the two age groups (<2 years and ≥2 years) and caregiver status. Caregivers (n = 117) identified numerous concerns and strengths across multiple categories. The most frequently reported concerns were related to aggressive behavior, language/communication, and sensory processing. The most frequently reported strengths were related to happiness, sociability, and love. The type of concerns and strengths reported were relatively consistent across age and caregiver status. These findings reinforce the value of caregivers' perspectives and offer a reminder to practitioners that infants/toddlers with PAE and their caregivers have many strengths that can be harnessed, in addition to a range of challenges that must be addressed.

Washington and Alaska statewide fetal alcohol spectrum disorder diagnostic clinical networks: Comparison of three decades of 4-Digit Code diagnostic outcomes and prenatal alcohol exposure histories.

Background: Fetal alcohol spectrum disorder (FASD) screening, diagnosis, intervention, research and prevention hinges on establishment of interdisciplinary FASD diagnostic clinics using an evidence-based method of diagnosis. In 1993 Washington State opened the first interdisciplinary FASD diagnostic clinic sponsored by the CDC as a FASD primary prevention study. Clinic data was used to develop the evidence-based FASD 4-Digit Diagnostic Code, paving the way for the clinic's expansion into a statewide network of FASD diagnostic clinics (Washington Fetal Alcohol Syndrome Diagnostic & Prevention Network), now in its 30th year. Alaska adopted this Washington model in 1999. Both states have also participated in the CDC Pregnancy Risk Assessment Monitoring System and Behavioral Risk Factor Surveillance System since the 1990s. Study objectives were to describe the two statewide FASD diagnostic networks; graphically compare the 4-Digit-Code FASD diagnoses and prenatal alcohol exposure (PAE) over 2-3 decades and illustrate how network data helped guide FASD public health policies and track successful prevention efforts. Methods: Retrospective descriptive study. Results: FASD diagnostic outcomes were similar across 2,532 Washington and 2,469 Alaskan patients. PAE in each State followed similar annual trajectories from 1991-2020. Both States documented significant decreases in FAS and PAE in the 1990s. Clinic data helped guide public health policies. Conclusions: Both States demonstrated the feasibility and value of establishing statewide interdisciplinary FASD diagnostic clinical networks using the FASD 4-Digit-Code. Legislative support, centralized data collection, and use of a single, evidence-based FASD diagnostic system have been key to the long-term, ongoing success of these two diagnostic networks.

Prevalence and patterns of sensory processing behaviors in a large clinical sample of children with prenatal alcohol exposure.

BACKGROUND: Atypical behavioral responses to sensation are reported in a large proportion of children affected by prenatal alcohol exposure (PAE). Systematic examination of symptoms across the fetal alcohol spectrum in a large clinical sample is needed to inform diagnosis and intervention. AIMS: To describe the prevalence and patterns of atypical sensory processing symptoms in a clinical sample of children with PAE. METHODS: Retrospective analysis of diagnostic clinical data from the University of Washington Fetal Alcohol Syndrome Diagnostic and Prevention Network (FASDPN). Participants were ages 3 through 11 years, had a diagnosis on the fetal alcohol spectrum, and Short Sensory Profile (SSP) assessment. The proportions of children categorized with definite differences on the SSP across selected clinical and demographic features were examined with chi-square analyses. OUTCOMES: The sample consisted of 325 children; 73.2 % had SSP total scores in the definite difference range. Atypical sensory processing symptoms were significantly more prevalent among children with higher reported levels of PAE. The prevalence of atypical symptoms was comparably high across age, levels of diagnostic severity, and other prenatal/postnatal risks. CONCLUSIONS: Results lend support for altered sensory processing as another domain of brain function affected by the teratogenic impact of PAE, guiding clinical work and research.

High facial specificity and positive predictive value are required to diagnose fetal alcohol syndrome when prenatal alcohol exposure is unknown.

BACKGROUND: Facial criteria with high specificity and positive predictive value (PPV) to prenatal alcohol exposure (PAE) are required to diagnose fetal alcohol syndrome (FAS) when documentation of PAE is unavailable. Not all fetal alcohol spectrum disorder (FASD) diagnostic guidelines appear to meet these criteria. METHODS: A dataset generated from a 10-year FAS screening of 1,602 children in fostercare conducted by the University of Washington FAS Diagnostic & Prevention Network was used to determine how well the FAS facial phenotype, microcephaly and growth deficiency (individually and in combination at varying levels of magnitude) predicted PAE. RESULTS: The 4-Digit-Code Rank 4 FAS facial phenotype was the only outcome that provided sufficient PPV and specificity to PAE (100%) to allow the facial phenotype to serve as confirmation of PAE in a diagnostic setting when PAE is unknown. Even minimal relaxation of the phenotype (e.g., Face Rank 3) resulted in PPV (35%) and specificity (88.7%) values too low to use as confirmation of PAE. Further relaxation of the facial criteria, as defined by the Hoyme et al., 2016 FASD guidelines, resulted in even lower PPV (17.9%) and specificity (76.6%); both too low to serve as confirmation of PAE in a diagnostic setting. The presence of all three physical features of FAS (Hoyme et al FAS facial phenotype, growth and OFC ≤10th percentile) did not increase PPV beyond chance (52%). CONCLUSIONS: FASD diagnostic guidelines that use relaxed criteria for the FAS facial phenotype risk misdiagnosing and over-diagnosing FAS and partial FAS when PAE is unknown.

Listening Difficulties in Children With Fetal Alcohol Spectrum Disorders: More Than a Problem of Audibility.

Purpose Data from standardized caregiver questionnaires indicate that children with fetal alcohol spectrum disorders (FASDs) frequently exhibit atypical auditory behaviors, including reduced responsivity to spoken stimuli. Another body of evidence suggests that prenatal alcohol exposure may result in auditory dysfunction involving loss of audibility (i.e., hearing loss) and/or impaired processing of clearly audible, "suprathreshold" sounds necessary for sound-in-noise listening. Yet, the nexus between atypical auditory behavior and underlying auditory dysfunction in children with FASDs remains largely unexplored. Method To investigate atypical auditory behaviors in FASDs and explore their potential physiological bases, we examined clinical data from 325 children diagnosed with FASDs at the University of Washington using the FASD 4-Digit Diagnostic Code. Atypical behaviors reported on the "auditory filtering" domain of the Short Sensory Profile were assessed to document their prevalence across FASD diagnoses and explore their relationship to reported hearing loss and/or central nervous system measures of cognition, attention, and language function that may indicate suprathreshold processing deficits. Results Atypical auditory behavior was reported among 80% of children with FASDs, a prevalence that did not vary by FASD diagnostic severity or hearing status but was positively correlated with attention-deficit/hyperactivity disorder. In contrast, hearing loss was documented in the clinical records of 40% of children with fetal alcohol syndrome (FAS; a diagnosis on the fetal alcohol spectrum characterized by central nervous system dysfunction, facial dysmorphia, and growth deficiency), 16-fold more prevalent than for those with less severe FASDs (2.4%). Reported hearing loss was significantly associated with physical features characteristic of FAS. Conclusion Children with FAS but not other FASDs may be at a particular risk for hearing loss. However, listening difficulties in the absence of hearing loss-presumably related to suprathreshold processing deficits-are prevalent across the entire fetal alcohol spectrum. The nature and impact of both listening difficulties and hearing loss in FASDs warrant further investigation.

Are Low-to-Moderate Average Alcohol Consumption and Isolated Episodes of Binge Drinking in Early Pregnancy Associated with Facial Features Related to Fetal Alcohol Syndrome in 5-Year-Old Children?

BACKGROUND: Fetal alcohol syndrome (FAS) typically is observed among individuals with high prenatal alcohol exposures (PAE), but exposure histories obtained in clinical diagnostic settings are often inaccurate. The present analysis used the Lifestyle During Pregnancy Study (LDPS) to assess the potential effects of low-to-moderate average weekly alcohol consumption and binge drinking in early pregnancy on facial features associated with FAS among children 5 years of age. METHODS: The analysis is a prospective follow-up study of 670 women and their children sampled from the LDPS cohort based on maternal alcohol consumption during pregnancy. The 4-Digit Code FAS Facial Photographic Analysis Software was used to measure the magnitude of expression of the 3 diagnostic facial features of FAS from standardized digital photographs. Logistic regression was used to estimate the odds of presenting with the FAS/partial fetal alcohol syndrome (PFAS) facial phenotypes relative to different patterns of prenatal alcohol exposure. RESULTS: Ten children presented with the FAS/PFAS facial phenotypes. None of the children sampled met the central nervous system (CNS) criteria for FAS or PFAS at age 5 years. All remained at risk for PFAS since some types of CNS dysfunction associated with this diagnosis may only be assessed at older ages. The FAS/PFAS facial phenotypes were 8.5-fold more likely among children exposed to an average of 1 to 4 drinks/wk and 2.5-fold more likely among children with a single binge exposure in gestational weeks 3 to 4 compared to children with no such exposures. The magnitude of expression of the FAS facial phenotype was significantly correlated with all other diagnostic features of FAS: growth deficiency, microcephaly, and measures of CNS dysfunction. CONCLUSIONS: These findings suggest that low-to-moderate levels of PAE or isolated binge exposures may place some fetuses at risk for FAS/PFAS. Thus, conservative advice is still for women to abstain from alcohol consumption during pregnancy.

Twin study confirms virtually identical prenatal alcohol exposures can lead to markedly different fetal alcohol spectrum disorder outcomes-fetal genetics influences fetal vulnerability.

BACKGROUND: Risk of fetal alcohol spectrum disorder (FASD) is not based solely on the timing and level of prenatal alcohol exposure (PAE). The effects of teratogens can be modified by genetic differences in fetal susceptibility and resistance. This is best illustrated in twins. OBJECTIVE: To compare the prevalence and magnitude of pairwise discordance in FASD diagnoses across monozygotic twins, dizygotic twins, full-siblings, and half-siblings sharing a common birth mother. METHODS: Data from the Fetal Alcohol Syndrome Diagnostic & Prevention Network clinical database was used. Sibling pairs were matched on age and PAE, raised together, and diagnosed by the same University of Washington interdisciplinary team using the FASD 4-Digit Code. This design sought to assess and isolate the role of genetics on fetal vulnerability/resistance to the teratogenic effects of PAE by eliminating or minimizing pairwise discordance in PAE and other prenatal/postnatal risk factors. RESULTS: As genetic relatedness between siblings decreased from 100% to 50% to 50% to 25% across the four groups (9 monozygotic, 39 dizygotic, 27 full-sibling and 9 half-sibling pairs, respectively), the prevalence of pairwise discordance in FASD diagnoses increased from 0% to 44% to 59% to 78%. Despite virtually identical PAE, 4 pairs of dizygotic twins had FASD diagnoses at opposite ends of the fetal alcohol spectrum-Partial Fetal Alcohol Syndrome versus Neurobehavioral Disorder/Alcohol-Exposed. CONCLUSION: Despite virtually identical PAE, fetuses can experience vastly different FASD outcomes. Thus, to protect all fetuses, especially the most genetically vulnerable, the only safe amount to drink is none at all.