The contribution of the Neurobehavioral Screening Tool to identifying fetal alcohol spectrum disorders in children at high risk of prenatal alcohol exposure and neurobehavioral deficits☆
Introduction
Alcohol is the most commonly used human teratogen, due to its availability, its abuse potential, and its economic value. In 1968, Lemoine et al. described the severe adverse effects of alcohol on prenatal development [1]. A few years later, Jones et al. described a pattern of malformations and developmental problems in children born to mothers with chronic alcoholism who consumed alcohol while pregnant [2]. This pattern, which was termed fetal alcohol syndrome (FAS), included typical facial dysmorphology and other physical malformations (i.e., cardiac, skeletal), developmental delay and growth retardation. Over the years, the deficits associated with prenatal exposure to alcohol became recognized as a spectrum, namely fetal alcohol spectrum disorders (FASD), with a range from mild to severe [3].
Epidemiological research demonstrates that FASD is a worldwide problem [4]. The recent estimated prevalence of FASD among school children in four US communities ranged from 1 to 5 %, while only up to 0.78 % of them were identified as having the complete syndrome (FAS) [5]. The evidence in Israel suggests extremely low rates of detection or diagnosis of FASD [6], [7]. However, according to the Organization for Economic Co-operation and Development, alcohol consumption in the general population of many countries including Israel is rising, particularly risky drinking behaviors (‘binge drinking’), and among young people and women [8]. In 2011, Senecky et al. described that 17.1 % of women who were surveyed about their drinking habits reported drinking alcohol during their pregnancy, while 0.8 % reported consuming a highly dangerous amount of at least four glasses of alcohol in one sitting (‘binge drinking’) [9]. In a similar survey conducted in 2020, 12.0 % of the women reported drinking alcohol during their current pregnancy, 1.1 % reported consuming more than two glasses of alcohol in one sitting and 28.1 % reported knowing other women who had consumed alcohol during pregnancy [10].
The diagnosis of FASD can be very challenging. First, it necessitates a verified history of prenatal alcohol exposure, which may be difficult to obtain. This is because complete and reliable information is often not available, especially for children who are adopted or in foster care. Second, the pathognomonic facial dysmorphology (flat upper lip, smooth philtrum and short palpebral fissures) associated with FAS does not appear in many children who are exposed to alcohol prenatally [11]. Thus, numerous children with FASD lack clear evidence of prenatal alcohol exposure and also pathognomonic facial features. These children often do not receive an FASD diagnosis or are diagnosed at a later stage [12], [13], and hence are not cared for or treated optimally.
FASD is characterized by complex neurobehavioral deficits [14], [15]. Thus, the majority of children with FASD exhibit disruptive behaviors, including aggression, impulsivity, rule breaking and asocial behavior [14], and regulation and conduct disorders [16]. These are in addition to their having lower IQ, learning disabilities and other cognitive deficits [17], [18], [19]. Early diagnosis and early intervention of FASD have been shown to be associated with long-term lessening of these symptoms [20]. On the other hand, lack of proper diagnosis or treatment may have negative consequences on children's function, future and role in society [21]; and negative effects on the coping ability of their families or caregivers [22]. Therefore, to improve the screening and diagnosis of FASD, efforts have been focused on identifying a distinct neurobehavioral profile – defined as the apparent expression (behavioral and developmental) of the central nervous system damage caused by prenatal alcohol exposure.
In 2006, Nash et al. described the Neurobehavioral Screening Tool (NST), a questionnaire to be filled by caregivers, requiring “yes or no” responses to each of 10 items [23]. The items were derived from the Achenbach Child Behavior Checklist. According to Nash et al., for children aged 6–16 years, answering “yes” to at least 6 of items 1 to 7 yielded a sensitivity of 86 % and specificity of 82 % for FASD. Other Canadian researchers have since sought to replicate the results in similar and wider age ranges, from 4 to 18 years. [24], [25], [26], [27]. In those studies, the NST demonstrated good sensitivity (63–98 %) but varying specificity (42–100 %), and was thus deemed suitable for screening purposes only and in need of further validation.
The purpose of the present study was to examine the contribution of the NST to identifying FASD in a cohort of children who were diagnosed with FASD, or who presented with other behavioral problems suspicious of FASD; and to compare these results to those of previous studies. To the best of our knowledge, this is the first examination of the NST in a non-Canadian cohort. In addition, we sought to identify other demographic, socioeconomic and behavioral characteristics associated with FASD, which could contribute to the diagnosis of children in risk of prenatal alcohol exposure.
Section snippets
Study population
The individuals included in this study were all examined in the Neurology and Child Developmental Clinic of our center during 2012–2020. This clinic serves as a national FASD clinic. Overall, 202 individuals were referred to the clinic for assessment of FASD due to their caregivers' or physicians' concern that they were exposed to alcohol prenatally, and that such exposure may have contributed to their behavioral problems. During their visits, caregivers were asked regarding their children's
Characteristics of the study population
The age range of the 151 children and young adults in the sample was 3.75 to 22 years. Males comprised 91 (60.2 %) of the cohort. Nineteen percent of the children and young adults were born in Israel; most of the remaining were born in ex-Soviet Union countries. The vast majority of the cohort were either adopted internationally [122 (80.8 %)] or adopted in Israel, and were in Israeli foster care [25 (16.6 %)]. Only 4 (2.6 %) were raised by their biological parents or family members. For most
Discussion
Our findings of 72.5 to 72.7 % sensitivity and 34.2 to 36.4 % specificity using the NST are lower than previously reported. This suggests lesser usefulness of NST as a screening tool, specifically among children at a high risk of prenatal alcohol exposure and neurobehavioral deficits. Nevertheless, our results show some effectiveness of the questionnaire in identifying individuals with definite diagnoses and individuals with confirmed prenatal alcohol exposure. The relatively low specificity
Conclusion
This report contributes to the growing literature in search of a valid neurobehavioral phenotype to facilitate the diagnosis and screening of FASD. In addition, this study highlights the everyday clinical challenges in diagnosing FASD in children in high-risk groups, and the necessity to develop a reliable screening and diagnostic tool. Identifying these children is important for provision of appropriate intervention such as to promote adaptive behavior and a supportive environment. Diagnosis
CRediT authorship contribution statement
All authors have made substantial contributions in the design, execution, and analysis of the paper.
Individual contribution is characterized by the following credits:
Dana Ronen – Conceptualization, Methodology, Resources, Validation, Formal analysis, Investigation, Writing - Original draft, Writing - Review & editing.
Yehuda Senecky MD – Supervision, Conceptualization, Methodology, Resources, Validation, Formal analysis, Investigation, Writing - Review & editing, Project administration.
Gabriel
Declaration of competing interest
None.
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Cited by (1)
Towards a Distinct Sleep and Behavioural Profile of Fetal Alcohol Spectrum Disorder (FASD): A Comparison between FASD, Autism and Typically Developing Children
2023, Journal of Integrative Neuroscience
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This work received no financial support.