Identifying the behavioural phenotype in Fetal Alcohol Spectrum Disorder: sensitivity, specificity and screening potential.

Psychology Department, The Hospital for Sick Children, Toronto, Cananda.
Archives of Women s Mental Health (Impact Factor: 2.16). 08/2006; 9(4):181-6. DOI: 10.1007/s00737-006-0130-3
Source: PubMed


In most cases of Fetal Alcohol Spectrum Disorder (FASD), the pathognomonic facial features are absent making diagnosis challenging, if not impossible, particularly when no history of maternal drinking is available. Also because FASD is often comorbid with Attention Deficit Hyperactivity Disorder (ADHD), children with FASD are frequently improperly diagnosed and receive the wrong treatment. Since access to psychological testing is typically limited or non-existent in remote areas, other diagnostic methods are needed to provide necessary interventions.
To determine if a characteristic behavioural phenotype distinguishes children with FASD from typically developing children and children with ADHD and use this information to create a screening tool for FASD diagnosis.
Parents and caregivers completed the Child Behavior Checklist (CBCL), a well-established standardized tool for evaluating children's behavioural problems. Results from 30 children with Fetal Alcohol Syndrome or Alcohol-Related Neurodevelopmental Disability, 30 children with ADHD, and 30 typically developing healthy children matched for age and socioeconomic status with FASD were analyzed. Based on our previous work, 12 CBCL items that significantly differentiated FASD and control groups were selected for further analyses. Stepwise discriminant function analysis identified behavioural characteristics most strongly differentiating groups and Receiver Operating Characteristics (ROC) curve analyses determined sensitivity and specificity of different item combinations.
Seven items reflecting hyperactivity, inattention, lying and cheating, lack of guilt, and disobedience significantly differentiated children with FASD from controls. ROC analyses showed scores of 6 or higher on these items differentiated groups with a sensitivity of 86%, specificity of 82%. For FASD and ADHD, two combinations of items significantly differentiated groups with high sensitivity and specificity (i) no guilt, cruelty, and acts young (sensitivity = 70%; specificity = 80% (ii) acts young, cruelty, no guilt, lying or cheating, steals from home, and steals outside (sensitivity = 81%; specificity = 72%). These items were used to construct a potential FASD screening tool.
Our findings identifying the behavioural characteristics differentiating children with FASD from typically developing children or children with ADHD have the potential for development of an empirically derived tool for FASD tool to be used in remote areas where psychological services are not readily available. This technique may speed up diagnosis and intervention for children without ready access to formal assessments.

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Available from: Joanne F Rovet, Dec 04, 2014
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    • "Their cognitive deficits include reduced levels of intelligence (Mattson, Riley, Gramling, Delis, & Jones, 1997) and memory (Rasmussen, Horne, & Witol, 2006; Willoughby, Sheard, Nash, & Rovet, 2008), as well as difficulties with attention (Coles, Platzman, Lynch, & Freides, 2002; Mattson, Calarco, & Lang, 2006) and executive functioning (Rasmussen, McAuley, & Andrew, 2007; Rasmussen, Wyper, & Talwar, 2009). Behavior problems reflect high rates of attention deficit hyperactivity disorder (ADHD; Nash et al., 2006; Rasmussen et al., 2010), conduct disorder (Nash, Koren, & Rovet, 2011) and autism traits (Bishop, Gahagan, & Lord, 2007; Stevens, Nash, Koren, & Rovet, 2012), while their social difficulties include inappropriate peer relationships (Keil, Paley, Frankel, & O'Connor, 2010; O'Connor et al., 2006), lack of social judgment (Kodituwakku, 2007), and poor social problem solving abilities (McGee, Bjorkquist, Price, Mattson, & Riley, 2009; McGee, Fryer, Bjorkquist, Mattson, & Riley, 2008; Stevens, Major, Rovet, & Desrocher, 2012). Research investigating the impact of exposure severity shows mixed results, with some studies describing poorer cognitive performance in children with FAS (Chasnoff, Wells, Telford, Schmidt, & Messer, 2010) and others showing children without the full FAS profile being at higher risk of social functioning deficits (Fast, Conry, & Loock, 1999; Schonfeld, Mattson, & Riley, 2005). "
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    ABSTRACT: Children with fetal alcohol spectrum disorders (FASD) show sociobehavioral impairments; however, the social cognitive profile contributing to these impairments is poorly understood. This study compared social perspective taking and empathy in children with FASD versus typically developing controls (TDC). Thirty-seven children with FASD and 21 TDC participated. Measures included parent-rated CBCL and SSIS, and NEPSY-II Theory of Mind, Test of Social Cognition and Index of Empathy. Parents rated the FASD group higher than TDC on indices of behavior problems and lower on indices of social skills and empathy. Children with FASD scored significantly below TDC on tasks requiring complex social cognition. The majority of correlations between social cognition and parent-ratings were not significant in FASD and TDC, with the exception of a negative correlation between self-reported empathy and parent-rated behavior difficulties in TDC. FASD subgroup analyses revealed lower theory of mind and empathy scores among children with ARND than pFAS/FAS. With regard to sex, males with FASD were rated as having more behavior difficulties than females, whereas TDC females obtained higher empathy ratings than males. In both groups, females scored higher on theory of mind and empathy indices. On theory of mind tasks, older children with FASD performed below younger, whereas younger TDC children performed more poorly than older. Children with FASD show reduced functioning on indices of sociobehavioral and social cognition, and the effects are influenced by sex and age. These findings provide insight into the clinical and social profile of children with FASD. (JINS, 2015, 21, 74-84)
    Journal of the International Neuropsychological Society 08/2015; 21(01):74-84. DOI:10.1017/S1355617714001088 · 2.96 Impact Factor
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    • "Attention difficulties arising from PEA continue into childhood (e.g., Lee et al., 2004; Kooistra et al., 2010). For example, children with PEA often meet criteria for ADHD based on clinical interviews (Koren et al., 2003; Fryer et al., 2007; Kooistra et al., 2010), score higher than same-aged peers on behavioral questionnaires that assess attention problems (Nanson and Hiscock, 1990; Brown et al., 1991; Coles et al., 1997; Lee et al., 2004; Nash et al., 2006; Astley et al., 2009), and are rated as more inattentive at school than children of mothers who did not (Brown et al., 1991). Yet, even as children with PEA consistently present with behavioural symptoms of inattention (e.g., Fryer et al., 2007), they do not always demonstrate deficits on experimental or clinical measures of attentional functioning. "
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    ABSTRACT: Parental reports of attention problems and clinical symptomatology of ADHD among children with fetal alcohol syndrome disorder (FASD) were assessed in relation to performance on standardized subtests of attentional control/shifting and selective attention from the Test of Everyday Attention for Children (TEA-Ch; Manly etal., 1998). The participants included 14 children with FASD with a mean chronological age (CA) of 11.7 years and a mean mental age (MA) of 9.7 years, and 14 typically developing (TD) children with no reported history of prenatal exposure to alcohol or attention problems with a mean CA of 8.4 years and a mean MA of 9.6 years. The children with FASD were rated by their caregivers as having clinically significant attention difficulties for their developmental age. The reported symptomatology for the majority of the children with FASD were consistent with a diagnosis of ADHD, combined type, and only one child had a score within the average range. These reports are consistent with the finding that the children with FASD demonstrated difficulties with attentional control/shifting, but inconsistent with the finding that they outperformed the TD children on a test assessing selective attention. These findings are considered within the context of the complexity in understanding attentional functioning among children with FASD and discrepancies across sources of information and components of attention.
    Frontiers in Human Neuroscience 03/2014; 8:119. DOI:10.3389/fnhum.2014.00119 · 3.63 Impact Factor
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    • "see Donaldson et al., 2011) as well as predisposition to mental health issues (Pei, Rinaldi, Rasmussen, Massey, & Massey, 2008). These include attention deficit disorder (Nash et al., 2006; O'Malley & Nanson, 2002; Rasmussen et al., 2010), conduct disorder (Nash, Koren, & Rovet, 2011), and autism (Bishop, Gahagan, & Lord, 2007; Stevens, Nash, Koren, & Rovet, 2012). As adults, individuals with FASD typically do not complete their educations and are unemployed while many experience depression, suicide risk, and trouble with the law (O'Malley & Huggins, 2005; Streissguth et al., 2004). "
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    ABSTRACT: Individuals diagnosed with alcohol-related neurodevelopmental disorder (ARND) exhibit difficulty on hippocampally mediated memory tasks and show reduced hippocampal size. However inconsistencies exist regarding the affected memory functions and where within the hippocampi effects occur. Given recent studies showing anterior and posterior segments support distinct memory functions and sex dimorphisms in hippocampal function, we asked whether these factors influence memory performance in youth with ARND (n = 18) and typically developing controls (n = 17). Participants received a battery of memory tests and a structural MRI scan. Right and left hippocampi were manually traced; anterior and posterior segments were delineated at the uncus. Measured were intracranial volumes (ICV) and right and left hippocampi and hippocampal segments. Volumes were adjusted for ICV. Relative to controls, the ARND group had lower IQs and memory performance on most tasks and marginally smaller ICVs. Left and right hippocampal volumes and posterior segments were smaller in the ARND group. Although no sex differences were observed between groups, females overall had larger anterior hippocampi than males. Positive and negative associations between hippocampal and selective memory indices were found in the ARND group only. These findings are the first to suggest that posterior hippocampal development may be compromised in youth with ARND. (JINS, 2014, 20, 181-191).
    Journal of the International Neuropsychological Society 02/2014; 20(2):181-91. DOI:10.1017/S1355617713001343 · 2.96 Impact Factor
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