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

Onesimo, Roberta; Delogu, Angelica Bibiana; Battista, Andrea; Leoni, Chiara; Veltri, Stefania; Rosa, Gabriella De; Zampino, Giuseppe

doi:10.1186/s13052-019-0759-y

Cited by 3 publications

(4 citation statements)

References 15 publications

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“…We were also unable to control for the exact date of diagnosis, which warrants future research as previous studies have acknowledged a strong association between early diagnosis of FASDs and fewer primary and/or secondary disabilities such as alcohol/drug problems, trouble with the law, and/or confinement 36 . Fetal alcohol biomarkers and genetic research are believed to play a central role in determining the onset of FASDs, however, due to the nature of our dataset, we were unable to control for related variables.…”

Section: Discussionmentioning

confidence: 90%

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

Kim

Jang

et al. 2020

Sci Rep

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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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Section: Discussionmentioning

confidence: 90%

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

Kim

Jang

et al. 2020

Sci Rep

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“…PAE is related not only to CHD but also to cardiac rhythm alterations in the absence of structural anomalies or cardiac channelopathies. Onesimo and colleagues reported two interesting cases of children affected by FASD according to Hoyme’s criteria [ 25 ]; the first case showed uniform and isolated premature ventricular contractions (PVCs), and the second case showed frequent premature atrial contractions (PACs) and short runs of ectopic atrial tachycardia [ 39 ]. Therefore, screening for arrhythmias in children affected by FASD without structural CHDs must be executed.…”

Section: Resultsmentioning

confidence: 99%

“…Significant correlations were described with ventricular and septal/atrial defects. Children exposed to alcohol in utero may display a 1.64-fold times increased risk of being affected by subtypes of conotruncal defects such as great artery transposition [ 26 , 39 , 40 ]. Both prenatal heavy drinking and binge drinking are strongly associated with a generally increased risk of having newborns with congenital heart defects [ 41 ].…”

Section: Introductionmentioning

confidence: 99%

Prenatal Alcohol Exposure and Metabolic Disorders in Pediatrics: The Role of the Oxidative Stress—A Review of the Literature

Derme,

Briante,

Ceccanti

et al. 2024

Children

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Prenatal alcohol exposure is responsible for increasing chronic disease risk in later life, including obesity and metabolic syndrome. Alcohol drinking may compromise endogenous antioxidant capacity, causing an increase in free radicals and reactive oxygen species in the newborn. Excessive reactive oxygen species could attack the cellular proteins, lipids, and nucleic acids, leading to cellular dysfunction. Moreover, oxidative stress could play a crucial role in the altered synthesis and release of neurotrophins and progressive mitochondrial modifications with uncontrolled apoptosis. This narrative review aims to underline the important role of alcohol abuse in oxidative stress events and consequent metabolic and neurocognitive impairments in children exposed to alcohol during gestational life.

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“…The baseline heart rate is a key non-invasive vital sign that can provide an early indication of distress or the need for follow-up (Acharya, Joseph, Kannathal, Lim, & Suri, 2006). When one author sought to identify the baseline heart rate in infants with PAE, the answers were inconclusive, with decreases (Oberlander et al, 2010) and increases (Onesimo et al, 2019) in heart rate being reported. Further, the baseline heart rate normally changes throughout the first year of life (Fleming et al, 2011).…”

Section: Introductionmentioning

confidence: 99%

Baseline heart rate in infants with prenatal alcohol exposure: A systematic review and independent analysis

McDonnell

Fornell

Ponce

et al. 2022

Birth Defects Research

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Background: Infants with fetal alcohol syndrome exhibit a range of developmental anomalies, many related to the heart (e.g., decreased heart rate variability). However, the baseline heart rate in this population remains unclear. We hypothesized that the age at which heart rate was measured or the age during exposure to alcohol affects the baseline heart rate. Methods: First, we conducted a systemic review to determine the published heart rate of infants with prenatal alcohol exposure (PAE). Exclusion criteria included potentially confounding factors, including the commonly associated phenotypes of small for gestational age and premature birth. Risk of bias was evaluated based on case study limitations, and data were compared with established heart rate norms. Then, we evaluated the precise age at heart rate measurement using existing datasets from the Collaborative Initiative on Fetal Alcohol Spectrum Disorders and the Maternal Lifestyle Study. Results: Based on the weighted means of six studies, the baseline heart rate was 4.6 bpm higher in infants with PAE (n = 253) than in control infants (n = 152). Using the individual patient data, baseline heart rates were similar between age-matched infants with PAE and control infants who were born full-term and showed no signs of growth restriction (ANOVA, p > .05; n = 49-124 infants per age and exposure).Conclusions: A systematic literature review suggested that heart rate is elevated in infants with PAE, but these findings are limited by the number of studies and how few studies included control infants. The analysis of individual patient data indicates that infants with PAE have normal baseline heart rates. This knowledge may help clinicians detect changes in cardiac function in infants with PAE.

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

Cited by 3 publications

References 15 publications

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

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

Prenatal Alcohol Exposure and Metabolic Disorders in Pediatrics: The Role of the Oxidative Stress—A Review of the Literature

Baseline heart rate in infants with prenatal alcohol exposure: A systematic review and independent analysis

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