Hospitalizations and mortality among patients with fetal alcohol spectrum disorders: a prospective study

Abstract With nearly 10% of women consuming alcohol during pregnancy, fetal alcohol spectrum disorders (FASDs) are becoming an increasing concern for clinicians and policymakers interested in the field of healthcare. Known as the range of mental and/or physical disabilities that occur among individuals with prenatal alcohol exposure, FASDs can result in dysmorphic features, problems with physical growth, neurobehavioral and cognitive problems that not only increase risk of various diseases, but also premature mortality. We investigated whether the diagnosis of FASDs result in increased risk of hospitalizations and mortality, with respect to FASD domains and relative diseases, when age effects are controlled for. The data for this study was taken from the National Health Insurance Service – National Sample Cohort (NHIS-NSC) between 2003 and 2013. The population attributable risk (PAR) statistic was used to estimate the percentage of hospitalizations and mortality attributable to FASDs and other factors. A time-dependent Cox proportional hazards model with age of diagnosis as the time-scale was employed to calculate adjusted hazard ratios and 95% CIs for hospitalizations and mortality among FASD populations compared to their general population peers. Among the 3,103 FASD cases, 27.5% experienced hospitalizations and 12.5% died. Overall, FASDs accounted for 853 FASD-attributable hospitalizations (51.0% of all hospitalizations in the study population) and 387 mortality events (34.5% of all deaths in the study population). 20.52% of hospitalizations and 21.35% of mortalities were attributable to FASDs in this population. Compared to the control group, FASD patients had a 1.25-fold (HR: 1.25, 95% CI: 1.05–1.49, p = 0.0114) increased risk of hospitalizations and a 1.33-fold (HR: 1.33, 95% CI: 1.07–1.67, p = 0.0118) increased risk of all-cause mortality. The most common cause for hospitalization was diseases of the nervous system, which accounted for 450 FASD-attributable hospitalizations (96.2% of all nervous system hospitalizations in the study population). In fact, FASD patients were 52 times more likely to be hospitalized for nervous system diseases than their peers (HR: 51.78, 95% CI: 29.09–92.17, p < .0001). The most common cause for mortality was neoplasms, which accounted for 94 FASD-attributable deaths (28.7% of all neoplasm deaths in the study population). However, FASD patients did not have increased risk of neoplasm mortality than the general population (HR: 0.88, 95% CI: 0.59–1.32, p < .0001). Overall, this study found that individuals diagnosed with FASDs have increased risk of both hospitalizations and mortality, compared to their general population peers. This is particularly so for diseases of the nervous system, which showed a 52-fold increase in hospitalizations and four-fold increase in mortality for FASD patients in our study. Likewise, while the association between FASDs and neoplasm mortality was not significant in our investigation, more attention by neurologists and related healthcare providers regarding the link between these two factors is necessary. Trial Registration: Institutional Review Board of Yonsei University’s Health System: Y-2019-0174.

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Association of adverse childhood experiences and neurodevelopmental disorders in people with fetal alcohol spectrum disorders (FASD) and non-FASD controls

BMC Pediatrics ◽

10.1186/s12887-019-1878-8 ◽

2019 ◽

Vol 19 (1) ◽

Cited By ~ 9

Author(s):

Cassondra Kambeitz ◽

Marilyn G. Klug ◽

Jacob Greenmyer ◽

Svetlana Popova ◽

Larry Burd

Keyword(s):

Neurodevelopmental Disorders ◽

Fetal Alcohol Spectrum Disorders ◽

Fetal Alcohol ◽

Cross Sectional ◽

Foster Home ◽

Childhood Experiences ◽

Adverse Childhood ◽

Increased Risk ◽

Spectrum Disorders ◽

Fetal Alcohol Spectrum

Abstract Background Fetal alcohol spectrum disorder (FASD) is a highly prevalent lifelong disorder with high rates of comorbid neurodevelopmental disorders. Individuals with FASD are often exposed to abuse, neglect and foster home placements which have uncertain effects on the lifelong course of FASD. In this study we compare the prevalence of adverse childhood events (ACEs) and neurodevelopmental disorders in subjects with fetal alcohol spectrum disorders (FASD) and non-FASD controls. Methods A cross-sectional chart review of patients referred to a regional developmental center was used to identify people with FASD and non-FASD controls. We recorded the number of ACEs and neurodevelopmental disorders in each patient’s chart. The most common diagnoses were attention deficit hyperactivity disorder, comprehension deficits, sleep disorders, and cognitive impairments. T-tests and a regression equation were utilized to determine significant differences between the groups. Results The review identified 203 subjects, 98 with FASD and 105 non-FASD controls. Group mean age was 8.6 years and 64.5% were male. People with FASD were more likely to have any ACEs (mean 5.3) with ACE scores 3.7 points higher than non-FASD controls (mean 1.69) (t = 11.29; p < .001). Increased ACEs were associated with increased rates of neurodevelopmental disorders for people with FASD (R = .179, p = .026) but not for non-FASD controls (R = .130, p = .094). Conclusions: Both FASD and subsequent exposure to ACEs are associated with increased risk for development of comorbid neurodevelopmental disorders. Prevention of ACEs during childhood may decrease risk for development of comorbid neurodevelopmental disorders.

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

Journal of Speech Language and Hearing Research ◽

10.1044/2018_jslhr-h-18-0359 ◽

2019 ◽

Vol 62 (5) ◽

pp. 1532-1548 ◽

Cited By ~ 1

Author(s):

Susan A. McLaughlin ◽

John C. Thorne ◽

Tracy Jirikowic ◽

Tiffany Waddington ◽

Adrian K. C. Lee ◽

...

Keyword(s):

Central Nervous System ◽

Nervous System ◽

Hearing Loss ◽

Fetal Alcohol Spectrum Disorders ◽

Fetal Alcohol ◽

Auditory Dysfunction ◽

Spectrum Disorders ◽

Processing Deficits ◽

Fetal Alcohol Spectrum ◽

Auditory Behavior

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.

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Accentuate the Negative: Grammatical Errors During Narrative Production as a Clinical Marker of Central Nervous System Abnormality in School-Aged Children With Fetal Alcohol Spectrum Disorders

Journal of Speech Language and Hearing Research ◽

10.1044/2017_jslhr-l-17-0128 ◽

2017 ◽

Vol 60 (12) ◽

pp. 3523-3537 ◽

Cited By ~ 4

Author(s):

John C. Thorne

Keyword(s):

Central Nervous System ◽

Nervous System ◽

Fetal Alcohol Spectrum Disorders ◽

Error Rates ◽

Fetal Alcohol ◽

Grammatical Errors ◽

Grammatical Error ◽

Spectrum Disorders ◽

Grammatical Complexity ◽

Fetal Alcohol Spectrum

Purpose The purpose of this study was to examine (a) whether increased grammatical error rates during a standardized narrative task are a more clinically useful marker of central nervous system abnormality in Fetal Alcohol Spectrum Disorders (FASD) than common measures of productivity or grammatical complexity and (b) whether combining the rate of grammatical errors with the rate of cohesive referencing errors can improve utility of a standardized narrative assessment task for FASD diagnosis. Method The method used was retrospective analysis of narrative and clinical data from 138 children (aged 7–12 years; 69 with FASD, 69 typically developing). Narrative analysis was conducted blind to diagnosis. Measures of grammatical error, productivity and complexity, and cohesion were used independently and in combination to predict whether a story was told by a child with an FASD diagnosis. Results Elevated grammatical error rates were more common in children with FASD, and this difference facilitated a more accurate prediction of FASD status than measures of productivity and grammatical complexity and, when combined with an accounting of cohesive referencing errors, significantly improved sensitivity to FASD over standard practice. Conclusion Grammatical error rates during a narrative are a viable behavioral marker of the kinds of central nervous system abnormality associated with prenatal alcohol exposure, having significant potential to contribute to the FASD diagnostic process.

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Cohesive Referencing Errors During Narrative Production as Clinical Evidence of Central Nervous System Abnormality in School-Aged Children With Fetal Alcohol Spectrum Disorders

American Journal of Speech-Language Pathology ◽

10.1044/2016_ajslp-15-0124 ◽

2016 ◽

Vol 25 (4) ◽

pp. 532-546 ◽

Cited By ~ 4

Author(s):

John C. Thorne ◽

Truman E. Coggins

Keyword(s):

Central Nervous System ◽

Nervous System ◽

Narrative Analysis ◽

Fetal Alcohol Spectrum Disorders ◽

Group Performance ◽

Alcohol Exposure ◽

Error Rates ◽

Fetal Alcohol ◽

Spectrum Disorders ◽

Fetal Alcohol Spectrum

Purpose Previous evidence suggests that cohesive referencing errors made during narratives may be a behavior that is revealing of underlying central nervous system abnormality in children with fetal alcohol spectrum disorders (FASD). The current research extends this evidence. Method Retrospective analysis of narrative and clinical data from 152 children (ages 6 to 14), 72 of whom had confirmed FASD, was used. Narrative analysis was conducted blind to diagnostic status, age, or gender. Group performance was compared. The associations between measures of cohesive referencing and clinically gathered indices of the degree of central nervous system abnormality were examined. Results Results show clear associations between elevated rates of cohesive referencing errors and central nervous system abnormality. Elevated error rates were more common in children with FASD than those without, and prevalence increased predictably across groups with more severe central nervous system abnormality. Risk is particularly elevated for those with microcephaly or a diagnosis of fetal alcohol syndrome. Conclusion Cohesive referencing errors during narrative are a viable behavioral marker of the kinds of central nervous system abnormality associated with prenatal alcohol exposure, having significant potential to become a valuable diagnostic and research tool.

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