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Executive Functions among Youth with Down Syndrome and Co-Existing Neurobehavioural Disorders

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Title: Executive Functions among Youth with Down Syndrome and Co-Existing Neurobehavioural Disorders
Language: English
Authors: Pritchard, A. E., Kalback, S., McCurdy, M.
Source: Journal of Intellectual Disability Research. Dec 2015 59(12):1130-1141.
Availability: Wiley-Blackwell. 350 Main Street, Malden, MA 02148. Tel: 800-835-6770; Tel: 781-388-8598; Fax: 781-388-8232; e-mail: cs-journals@wiley.com; Web site: http://www.wiley.com/WileyCDA
Peer Reviewed: Y
Page Count: 12
Publication Date: 2015
Document Type: Journal Articles
Reports - Research
Descriptors: Executive Function, Down Syndrome, Neurological Impairments, Behavior Problems, Comorbidity, Behavior Rating Scales, Psychometrics, Autism, Pervasive Developmental Disorders, Factor Analysis, Metacognition, Inhibition, Intellectual Disability
DOI: 10.1111/jir.12217
ISSN: 0964-2633
Abstract: Background: Executive function (EF) deficits are :a recognised component of the cognitive phenotype of youth with Down Syndrome (DS). Recent research in this area emphasises the use of behaviour ratings, such as the Behavior Rating Inventory of Executive Functions-Preschool Version (BRIEF-P), to capture the real-world applications of executive functions. To account for the intellectual functioning of youth with DS, this measure is used out of age range; however, its psychometric properties when used in this fashion are unknown. The goals of this study are to evaluate psychometric characteristics of the BRIEF-P among youth with DS and to examine the pattern of EF strengths/weaknesses in children with DS and co-occurring psychiatric conditions. Method: A total of 188 clinically referred youth with DS, ages 3-13 were rated by their caregivers using the BRIEF-P. These youth were evaluated by a clinician with expertise in DS and were characterised as having no co-occurring behavioural disorder (Typical DS group), co-occurring Autism Spectrum Disorder (DS?+?ASD) or co-occurring Disruptive Behaviour Disorder (DS?+?DBD). Results: An exploratory factor analysis of item-level BRIEF-P data from clinically referred youth with DS supported the theoretically derived three-factor structure originally proposed for the BRIEF-P (Emergent Metacognition, Flexibility and Inhibitory Self-Control); however, the item composition of each factor varied somewhat in comparison to the original structure of the measure. Group comparisons indicate that, while youth with typical DS evidence fewer executive function difficulties across all domains, youth with DS?+?ASD show the greatest weaknesses in Emergent Metacognition, and youth with DS?+?DBD show significant difficulties in both Emergent Metacognition and Inhibition. Conclusions: These findings offer preliminary support for use of the BRIEF-P with clinically referred youth with Down Syndrome. Some scoring modifications may be necessary if the theoretically derived index scores are to be used with this population. BRIEF-P scores may offer an empirical basis for differentiating DS youth with varying behavioural profiles.
Abstractor: As Provided
Entry Date: 2016
Accession Number: EJ1087751
Database: ERIC
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  Value: <anid>AN0111334429;eul01dec.15;2018Jul06.12:42;v2.2.500</anid> <title id="AN0111334429-1">Executive functions among youth with Down Syndrome and co-existing neurobehavioural disorders. </title> <p>Background: Executive function (EF) deficits are a recognised component of the cognitive phenotype of youth with Down Syndrome (DS). Recent research in this area emphasises the use of behaviour ratings, such as the Behavior Rating Inventory of Executive Functions—Preschool Version (BRIEF‐P), to capture the real‐world applications of executive functions. To account for the intellectual functioning of youth with DS, this measure is used out of age range; however, its psychometric properties when used in this fashion are unknown. The goals of this study are to evaluate psychometric characteristics of the BRIEF‐P among youth with DS and to examine the pattern of EF strengths/weaknesses in children with DS and co‐occurring psychiatric conditions. Method: A total of 188 clinically referred youth with DS, ages 3–13 were rated by their caregivers using the BRIEF‐P. These youth were evaluated by a clinician with expertise in DS and were characterised as having no co‐occurring behavioural disorder (Typical DS group), co‐occurring Autism Spectrum Disorder (DS + ASD) or co‐occurring Disruptive Behaviour Disorder (DS + DBD). Results: An exploratory factor analysis of item‐level BRIEF‐P data from clinically referred youth with DS supported the theoretically derived three‐factor structure originally proposed for the BRIEF‐P (Emergent Metacognition, Flexibility and Inhibitory Self‐Control); however, the item composition of each factor varied somewhat in comparison to the original structure of the measure. Group comparisons indicate that, while youth with typical DS evidence fewer executive function difficulties across all domains, youth with DS + ASD show the greatest weaknesses in Emergent Metacognition, and youth with DS + DBD show significant difficulties in both Emergent Metacognition and Inhibition. Conclusions: These findings offer preliminary support for use of the BRIEF‐P with clinically referred youth with Down Syndrome. Some scoring modifications may be necessary if the theoretically derived index scores are to be used with this population. BRIEF‐P scores may offer an empirical basis for differentiating DS youth with varying behavioural profiles. © 2015 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd</p> <p>ADHD; autism; behavioral phenotyppes; down syndrome; intellectual disability; mental health; psychiatric disorders</p> <p>The cognitive phenotype of individuals with Down Syndrome (DS) is characterised by mild to moderate intellectual disability(ID) (Hodapp et al. [<reflink idref="bib23" id="ref1">23</reflink>] ; Silverman [<reflink idref="bib40" id="ref2">40</reflink>] ), stronger visual–perceptual and nonverbal communication skills (Jarrold et al. [<reflink idref="bib25" id="ref3">25</reflink>] ;Fidler et al. [<reflink idref="bib17" id="ref4">17</reflink>] ), weaker expressive language skills, with somewhat stronger receptive language skills (Rondal & Comblain [<reflink idref="bib35" id="ref5">35</reflink>] ; Sigman & Ruskin [<reflink idref="bib39" id="ref6">39</reflink>] ; Chapman & Hesketh [<reflink idref="bib7" id="ref7">7</reflink>] ), and difficulties with motor planning, coordination and speed (Mon‐Williams et al. [<reflink idref="bib32" id="ref8">32</reflink>] ; Latash [<reflink idref="bib30" id="ref9">30</reflink>] ).</p> <p>In addition, deficits in executive functions (EF) are now well established. Executive functions are defined as the dynamic self‐regulatory system that organises and directs cognitive, behavioural and emotional activity (Denckla [<reflink idref="bib12" id="ref10">12</reflink>] ; Pennington & Ozonoff [<reflink idref="bib34" id="ref11">34</reflink>] ). Among adults with DS, impairment in the specific EF areas of inhibitory control (Kittler et al. 2006), set‐shifting, sustained attention and planning (Rowe et al. [<reflink idref="bib37" id="ref12">37</reflink>] ), as well as flexibility and memory (Costanzo et al. 2013), have been identified. A similar pattern of EF deficits has been found among children and adolescents with DS, when compared with mental age‐matched peers (Lanfranchi et al. [<reflink idref="bib29" id="ref13">29</reflink>] ). Working memory difficulties, in particular, have been repeatedly identified in youth with DS (Baddeley & Jarrold [<reflink idref="bib2" id="ref14">2</reflink>] ; Kogan et al. [<reflink idref="bib28" id="ref15">28</reflink>] ).</p> <p>These EF deficits were captured by performance‐based measures, but disagreement remains regarding the utility of performance‐based measures of EF relative to parent or teacher ratings of the construct, particularly in terms of the ecological validity of the measures relative to symptoms that impact youth in daily life. There is mounting evidence that performance‐based measures and behaviour ratings of EF do not assess the same construct (Toplak et al. [<reflink idref="bib42" id="ref16">42</reflink>] ), but that they might provide complementary information (Isquith et al. [<reflink idref="bib24" id="ref17">24</reflink>] ). While performance‐based EF measures typically assess specific, individual executive functions under highly structured and standardised conditions (Toplak et al. [<reflink idref="bib42" id="ref18">42</reflink>] ), rating scales of EF, such as the Behavior Rating Inventory of Executive Function (BRIEF; Gioia et al. [<reflink idref="bib21" id="ref19">21</reflink>] ), were developed to tap into complex, real‐world manifestations of executive skills (Roth et al. [<reflink idref="bib36" id="ref20">36</reflink>] ), which are at least equally important for adaptive functions. The only studies to date that have examined the use of ratings‐based EF measures among youth with DS have utilised the Behavior Rating Inventory of Executive Function, Preschool version (BRIEF‐P; Gioia et al. [<reflink idref="bib22" id="ref21">22</reflink>] ), which was developed to assess functional EF skills among typically developing preschool‐aged children. Considering the developmental delays and associated adaptive skill deficits in children and adolescents with DS (Van Duijn et al. [<reflink idref="bib43" id="ref22">43</reflink>] ), the BRIEF‐P is believed to contain a more relevant sample of developmentally appropriate behaviours for this particular population. The BRIEF‐P has therefore been used outside of the age range upon which it was normed in an effort to evaluate the executive functions of youth with DS.</p> <p>Lee et al. ([<reflink idref="bib31" id="ref23">31</reflink>] ) evaluated 26 young children with DS using the BRIEF‐P parent rating and found that these children evidenced deficits in the areas of working memory and planning/organisation, relative to the normative sample. Daunhauer et al. ([<reflink idref="bib11" id="ref24">11</reflink>] ) compared 25 children with DS (ages 5–11) to 23 mental age‐matched typically developing children on BRIEF‐P parent and teacher ratings. They found significant deficits among the DS group in both teacher‐ and parent‐reported working memory and planning/organisation, and in parent‐reported inhibitory control.</p> <p>While these findings are promising, there is currently no information available regarding the psychometric properties of the BRIEF‐P in youth with DS. Studies of youth with DS have used the measure out of age range to account for the intellectually disabled status of the participants; however, it is not yet known whether the BRIEF‐P functions similarly in this group. The first goal of the present study is to evaluate the psychometric characteristics of the BRIEF‐P among clinically referred youth with DS.</p> <hd id="AN0111334429-2">Executive function deficits and maladaptive behaviour</hd> <p>While many youth with DS evidence a profile characterised by relatively fewer behavioural and social difficulties than their intellectually disabled peers without DS (Chapman & Hesketh [<reflink idref="bib7" id="ref25">7</reflink>] ; Fidler [<reflink idref="bib16" id="ref26">16</reflink>] ), they remain at increased risk, relative to typically developing youth, for the development of psychopathology, with estimates ranging from 18 to 38% of DS youth manifesting a co‐occurring psychiatric condition (Gath & Gumley [<reflink idref="bib18" id="ref27">18</reflink>] ; Gillberg & Gillberg [<reflink idref="bib19" id="ref28">19</reflink>] ; Myers & Pueschel [<reflink idref="bib33" id="ref29">33</reflink>] ; Coe et al. [<reflink idref="bib9" id="ref30">9</reflink>] ; Dykens, [<reflink idref="bib15" id="ref31">15</reflink>] ). Among children without DS, a positive relationship between executive functions and adaptive behaviour has been demonstrated (Clark et al. [<reflink idref="bib8" id="ref32">8</reflink>] ), and it is obvious how dysfunction in the executive system, such as difficulties with emotional regulation or inhibitory control, might give rise to maladaptive behaviours. While relatively little is known about the EF profiles of children with DS, even less is understood about the ways in which co‐occurring maladaptive behaviour or psychiatric disorder is associated with EF in this population.</p> <p>Many children with DS who demonstrate maladaptive behaviours meet criteria for one or more psychiatric diagnoses (Myers & Pueschel [<reflink idref="bib33" id="ref33">33</reflink>] ; Capone et al. [<reflink idref="bib5" id="ref34">5</reflink>] ; Capone et al. [<reflink idref="bib6" id="ref35">6</reflink>] ). However, the clinical formulation for arriving at a psychiatric diagnosis in children with ID is less straightforward because the clinician must attempt to apply criteria that were developed primarily for children without ID (Dykens [<reflink idref="bib14" id="ref36">14</reflink>] ). The use of standardised rating scales to systematically assess a wide range of behaviours represents a more rigorous approach to diagnosis (Dykens [<reflink idref="bib14" id="ref37">14</reflink>] ). Therefore, a second goal of this study is to examine the pattern of EF strengths and weaknesses across youth with DS who have co‐occurring psychiatric conditions, in comparison with ‘typical’ youth with DS.</p> <hd id="AN0111334429-3">Methods</hd> <hd id="AN0111334429-4">Participants</hd> <p>This study's participants were ascertained sequentially through initial visits to the Down Syndrome Clinic at the Kennedy Krieger Institute between 2004 and 2012. During this period, 258 individuals, ages 3–13, with and without behaviour problems, were evaluated by a single clinician (gtc). Clinic patients' caregivers were asked for written informed consent for the use of data from their medical record for this study. Of the children treated in the clinic during this timeframe, 92% were enrolled in the study, and 86% had complete BRIEF‐P data. Participants were not excluded on the basis of previous or current medical conditions, medication use, level of ID or demographic variables other than age. We analysed 183 completed BRIEF‐P rating scales representing children with Typical DS (n = 24), DS with co‐occurring Autism Spectrum Disorder (DS + ASD; n = 61) and DS with co‐occurring Disruptive Behaviour Disorder (DS + DBD; n = 98). Participants who did not meet the above criteria for ASD or DBD, but who demonstrated Stereotypic Movement Disorder (SMD; n = 39) without significant impairment in social reciprocity, were excluded from the sample because of lack of diagnostic specificity per ( Ji et al. [<reflink idref="bib26" id="ref38">26</reflink>] ) study. Per karyotype reports, the majority of participants (86%) had documented trisomy 21, while 3% had complete Robertsonian translocation, and 2% had mosaicism. Karyotype analysis was unavailable on 9% of participants who had phenotypic features consistent with DS.</p> <p>The average age for the full sample was 6.99 years (SD = 2.47). No significant differences between groups on age were found (F(<reflink idref="bib2" id="ref39">2</reflink>, 192) = 1.89, P = .154, η<sups>2</sups> = .019). Group‐specific age information is as follows: DS + ASD mean age of 6.52 years (SD = 2.49, range = 3–12), Typical DS mean age of 6.96 years (SD = 2.23, range = 3–13) and DS + DBD mean age of 7.28 years (SD = 2.50, range = 3–12).</p> <p>The clinic‐based nature of the sample utilised in the present study offers both advantages and disadvantages. While the present sample affords the opportunity to examine EF among individuals with a broader range of behavioural difficulties than might characterise the population of youth with DS, it also limits the potential generalisability of our findings regarding the psychometric properties of the BRIEF‐P.</p> <hd id="AN0111334429-5">Procedures</hd> <p>Approval for this study was granted by the local Institutional Review Board.</p> <hd id="AN0111334429-6">DSM‐IV‐TR classification</hd> <p>Participants classified as Typical DS did not manifest significant behavioural concerns as reported by their parents and confirmed by the examining physician; thus no DSM‐IV assignment was made. Participants presenting with behavioural concerns as determined by the parents or examining physician were assigned a primary diagnosis according to DSM‐IV‐TR criteria (American Psychiatric Association [<reflink idref="bib1" id="ref40">1</reflink>] ) using the information obtained from caretakers during the clinical evaluation, behavioural observations during unstructured play and in interaction with parents and the examiner. A single evaluator (gtc) completed a checklist of DSM‐IV‐TR criteria following the evaluation and rated all participants. A primary diagnosis was determined based on the predominant and most functionally impairing behavioural features present during the past 6 months. Each participant was given a primary diagnosis only, operationalised to be parsimonious while assigning children into mutually exclusive categories. Participants classified with Autism Spectrum Disorder were defined as children who met DSM‐IV‐TR diagnostic criteria for Autism (299.00), Pervasive Developmental Disorder—Not Otherwise Specified (299.80) or late‐onset Autism (299.10). Participants classified with Disruptive Behaviour Disorder met DSM‐IV‐TR criteria for either Oppositional Defiant Disorder (313.81), Disruptive Behaviour Disorder—Not Otherwise Specified (312.90) and/or Attention Deficit/Hyperactivity Disorder (314.01), without also meeting criteria for ASD.</p> <hd id="AN0111334429-7">Measures</hd> <hd id="AN0111334429-8">Behavioural Rating Inventory of Executive Function‐Preschool Version (BRIEF‐P)</hd> <p>The BRIEF‐P is a behaviour rating measure of everyday/functional EF skills in a preschool age population of typically developing children (Gioia et al. [<reflink idref="bib22" id="ref41">22</reflink>] ). The BRIEF‐P contains 63 items falling on five theoretically derived subscales (Inhibit, Shift, Emotional Control, Working Memory and Plan/Organise), each reflective of a specific aspect of executive function. Three composite indices, as well as a Global Executive Composite, are based on combinations of subscale scores. The Inhibitory Self‐Control Index (26 items) is comprised of the Inhibit and Emotional Control subscales, the Flexibility Index (20 items) of the Shift and Emotional Control subscales and the Emergent Metacognition Index (27 items) of the Working Memory and Plan/Organise subscales. The Global Executive Composite incorporates information derived from all five subscales. The subscales have good internal consistency (Chronbach's alpha = .80–.97) and test–retest reliability (rs = .78–.90; (Sherman & Brooks, [<reflink idref="bib38" id="ref42">38</reflink>] )). Primary caregivers were asked to endorse the extent to which they observe behaviours corresponding to particular executive functions using a 3‐point Likert scale (1 = never, 2 = sometimes, 3 = often).</p> <hd id="AN0111334429-9">General cognitive function</hd> <p>Level of cognitive functioning was determined by review of existing psychological reports generated within one year of the participant's initial evaluation in the Down Syndrome Clinic. We were able to obtain estimates of cognitive functioning in 43% of the participants with BRIEF‐P behaviour ratings, including 21 participants from the autism spectrum group (34%), 51 from the disruptive behaviour group (47%) and 11 from the Typical DS group (44%).</p> <p>Because of the paucity of norm‐referenced IQ measures developed for use with very low‐functioning children and adolescents, these youth are sometimes unable to accurately complete even the easiest items on IQ tests designed for children their age. For these youth, in order to gain an estimate of their abilities, which cannot be accurately estimated using age‐appropriate IQ tests, measures of developmental or intellectual functioning designed and normed for younger children are administered. In the present sample, 59 participants (71% of the 83 with IQ estimates) completed age‐appropriate IQ measures, while 24 (29% of those with IQ scores) completed measures of developmental/intellectual functioning normed for younger children. For the latter children, an age equivalent was calculated based on their performance on the measure administered, then a ratio IQ score was estimated by dividing the calculated age equivalent by their chronological age. For the remainder of individuals within the sample (n = 59), norm‐referenced IQ scores were calculated based on the age‐appropriate measures administered.</p> <p>The proportion of participants who received an age‐appropriate, norm referenced intelligence test (e.g. Wechsler Intelligence Scale for Children) varied by clinical grouping, with 10 (91%) of the Typical DS group, 8 (38%) of the autism spectrum group and 41 (80%) of the disruptive group receiving a norm‐referenced IQ score. The group of participants who completed an age‐appropriate intelligence test earned significantly higher scores (M = 50.51, SD = 10.34, range = 28–75) than the participants who completed a measure normed for younger children (M = 40, SD = 20.33, range = 16–88; t(<reflink idref="bib79" id="ref43">79</reflink>) = 3.08, P = .003); however, these two groups' IQ estimate scores are not entirely comparable because of their discrepant calculation methods.</p> <p>A chi‐squared test of independence indicated that the proportion of cognitively higher‐functioning youth was significantly larger in the Typical DS and DS + DBD groups, as compared with the DS + ASD group (χ<sups>2</sups>(<reflink idref="bib2" id="ref44">2</reflink>) = 15.37, P < .000, ϕ = .43).</p> <p>Because the cognitive data were collected by various examiners using different instruments across dissimilar settings, findings associated with these data should be considered preliminary at this time.</p> <hd id="AN0111334429-10">Data analysis plan</hd> <p>Exploratory factor analysis (EFA) was conducted to investigate the factor structure of the BRIEF‐P in a mixed‐age group of clinically referred children with Down Syndrome. Maximum likelihood estimation was used as the extraction method, and all derived factors were subjected to promax rotation in order to account for their anticipated oblique nature. On the basis of the factor analyses findings, BRIEF‐P subscale scores were calculated by averaging the scores of the items loading on each factor; thus the three BRIEF‐P subscales that were derived from the EFA represent the mean of unweighted raw scores.</p> <p>The EFA‐derived BRIEF‐P subscale scores were then compared across clinically based groups of children with Down Syndrome using MANOVA with follow‐up ANOVAs and Bonferroni‐corrected post hoc tests.</p> <p>Finally, differences in BRIEF‐P subscale scores between the relatively higher functioning group of children with norm‐referenced IQ scores and the relatively lower functioning group of children with ratio‐calculated IQ scores were evaluated using a series of independent samples t‐tests. Further, among the subgroup of participants for whom a norm‐referenced estimate of cognitive functioning was available, the relationship between IQ scores and BRIEF‐P subscale scores was examined with a series of Pearson correlations.</p> <hd id="AN0111334429-11">Results</hd> <hd id="AN0111334429-12">Exploratory factor analysis</hd> <p>Based on both conceptual and statistical considerations, exploratory factor analysis solutions involving between two and five factors were examined for the BRIEF‐P. Solutions involving fewer than two factors were overly heterogeneous conceptually, while those involving more than five factors offered distinctions that were too fine‐grained to be clinically useful. Examination of the scree plot indicated ‘bends’ at the 2<sups>nd</sups> and 5<sups>th</sups> factors, further supporting comparison of the two‐ through five‐factor solutions. Bayesian Information Criterion (BIC), Akaike Information Criterion (AIC) and χ<sups>2</sups> values were calculated as estimates of fit for each factor solution. Factor solutions that are more parsimonious and offer a better fit for the data will demonstrate smaller BIC, AIC and χ<sups>2</sups> values. A comparison of these values across the factor solutions (Table [NaN] ) suggests that, as we move from a two‐ to a five‐factor solution, both χ<sups>2</sups> and AIC values systematically decrease; however, the BIC value is smallest for the three‐factor solution. The number of items that were dropped because of cross‐loading or loadings less than.400 were minimised in the three‐factor solution (15 items dropped, as compared with 17 items dropped from the two‐factor solution, 19 items from the four‐factor solution and 22 items from the five‐factor solution). Therefore, the three‐factor solution was determined to be the most parsimonious while still allowing for clinically useful distinctions to be made. The 3‐factor solution included factors characterised as: Emergent Metacognition (aspects of cognitive problem‐solving, learning and memory), Flexibility (aspects of emotion regulation and behavioural flexibility) and Inhibition (aspects of response inhibition and motor activity). Of the total variance accounted for by the three‐factor solution, the Emergent Metacognition factor comprised 44%, the Flexibility factor comprised 30% and the Inhibition factor comprised 25%.</p> <p>Chi‐square, AIC and BIC values across factor solutions</p> <p> <ephtml> <table><tr><th id="jir12217-ent-0001" align="left">Number of factors</th><th id="jir12217-ent-0002" align="left">X<sup>2</sup></th><th id="jir12217-ent-0003" align="left">AIC</th><th id="jir12217-ent-0004" align="left">BIC</th></tr><tr><td id="jir12217-ent-0005" align="left">2</td><td id="jir12217-ent-0006" align="char" char=".">3165.23</td><td id="jir12217-ent-0007" align="char" char=".">3885.78</td><td id="jir12217-ent-0008" align="char" char=".">4284.2</td></tr><tr><td id="jir12217-ent-0009" align="left">3</td><td id="jir12217-ent-0010" align="char" char=".">2718.22</td><td id="jir12217-ent-0011" align="char" char=".">3507.73</td><td id="jir12217-ent-0012" align="char" char=".">4100.59</td></tr><tr><td id="jir12217-ent-0013" align="left">4</td><td id="jir12217-ent-0014" align="char" char=".">2489.98</td><td id="jir12217-ent-0015" align="char" char=".">3376.83</td><td id="jir12217-ent-0016" align="char" char=".">4160.93</td></tr><tr><td id="jir12217-ent-0017" align="left">5</td><td id="jir12217-ent-0018" align="char" char=".">2307.10</td><td id="jir12217-ent-0019" align="char" char=".">3294.54</td><td id="jir12217-ent-0020" align="char" char=".">4266.69</td></tr></table> </ephtml> </p> <p>1 Note. Lower X2 statistics and AIC and BIC values indicate a better/more parsimonious fit.</p> <p>Factor loadings for the three‐factor solution are presented in Table [NaN] . Items loading less than.400 on all subscales (items 3, 8, 13, 22, 24, 25, 35, 41, 47, 48, 50, 53, 55, 56 and 60) were deleted. Based on the three‐factor solution, BRIEF‐P subscales were calculated for use in all remaining analyses as the unweighted mean of the items loading on a given factor, in order to avoid capitalising on chance aspects of the present sample.</p> <p>Factor loadings for the 3‐factor solution</p> <p> <ephtml> <table><tr><th id="jir12217-ent-0021" align="left">Item</th><th align="left" id="jir12217-ent-0022">Factor</th></tr><tr><th align="left" id="jir12217-ent-0024">Emergent metacog.</th><th align="left" id="jir12217-ent-0025">Flexibility</th><th align="left" id="jir12217-ent-0026">Inhibition</th></tr><tr><td id="jir12217-ent-0027" align="left">1. Overreacts to small problems</td><td align="left" id="jir12217-ent-0028" /><td align="char" char="." id="jir12217-ent-0029">.622</td><td id="jir12217-ent-0030" /></tr><tr><td id="jir12217-ent-0031" align="left">2. When given two things to do, remembers only the first or last</td><td id="jir12217-ent-0032" align="left">.631</td><td id="jir12217-ent-0033" /><td id="jir12217-ent-0034" /></tr><tr><td id="jir12217-ent-0035" align="left">3. Is unaware of how his/her behaviour affects or bothers others</td><td id="jir12217-ent-0036" align="left">—</td><td id="jir12217-ent-0037" align="left">—</td><td id="jir12217-ent-0038" align="left">—</td></tr><tr><td id="jir12217-ent-0039" align="left">4. When instructed to clean up, puts things away in a disorganised way</td><td id="jir12217-ent-0040" align="left">.562</td><td id="jir12217-ent-0041" /><td id="jir12217-ent-0042" /></tr><tr><td id="jir12217-ent-0043" align="left">5. Becomes upset with new situations</td><td align="left" id="jir12217-ent-0044" /><td align="char" char="." id="jir12217-ent-0045">.649</td><td id="jir12217-ent-0046" /></tr><tr><td align="left" id="jir12217-ent-0047">6. Has explosive, angry outbursts</td><td align="left" id="jir12217-ent-0048" /><td align="char" char="." id="jir12217-ent-0049">.729</td><td id="jir12217-ent-0050" /></tr><tr><td align="left" id="jir12217-ent-0051">7. Has trouble carrying out actions needed to complete tasks</td><td id="jir12217-ent-0052" align="left">.716</td><td id="jir12217-ent-0053" /><td id="jir12217-ent-0054" /></tr><tr><td align="left" id="jir12217-ent-0055">8. Does not stop laughing at funny things/events when others stop</td><td id="jir12217-ent-0056" align="left">—</td><td id="jir12217-ent-0057" align="left">—</td><td id="jir12217-ent-0058" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0059">9. Needs to be told to begin a task even when willing to do it</td><td id="jir12217-ent-0060" align="left">.652</td><td id="jir12217-ent-0061" /><td id="jir12217-ent-0062" /></tr><tr><td align="left" id="jir12217-ent-0063">10. Has trouble adjusting to new people</td><td align="left" id="jir12217-ent-0064" /><td align="char" char="." id="jir12217-ent-0065">.557</td><td id="jir12217-ent-0066" /></tr><tr><td align="left" id="jir12217-ent-0067">11. Becomes upset too easily</td><td align="left" id="jir12217-ent-0068" /><td align="char" char="." id="jir12217-ent-0069">.722</td><td id="jir12217-ent-0070" /></tr><tr><td align="left" id="jir12217-ent-0071">12. Has trouble concentrating on games, puzzles or play activities</td><td id="jir12217-ent-0072" align="left">.670</td><td id="jir12217-ent-0073" /><td id="jir12217-ent-0074" /></tr><tr><td align="left" id="jir12217-ent-0075">13. Has to be more closely supervised than similar playmates</td><td id="jir12217-ent-0076" align="left">—</td><td id="jir12217-ent-0077" align="left">—</td><td id="jir12217-ent-0078" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0079">14. When sent to get something, forgets what he/she is supposed to get</td><td id="jir12217-ent-0080" align="left">.620</td><td id="jir12217-ent-0081" /><td id="jir12217-ent-0082" /></tr><tr><td align="left" id="jir12217-ent-0083">15. Is upset by change in plans or routine</td><td align="left" id="jir12217-ent-0084" /><td align="char" char="." id="jir12217-ent-0085">.603</td><td id="jir12217-ent-0086" /></tr><tr><td align="left" id="jir12217-ent-0087">16. Has outbursts for little reason</td><td align="left" id="jir12217-ent-0088" /><td align="char" char="." id="jir12217-ent-0089">.707</td><td id="jir12217-ent-0090" /></tr><tr><td align="left" id="jir12217-ent-0091">17. Repeats the same mistakes over and over even after help is given</td><td id="jir12217-ent-0092" align="left">.661</td><td id="jir12217-ent-0093" /><td id="jir12217-ent-0094" /></tr><tr><td align="left" id="jir12217-ent-0095">18. Acts wilder or sillier than others in groups</td><td align="left" id="jir12217-ent-0096" /><td id="jir12217-ent-0097" /><td align="char" char="." id="jir12217-ent-0098">.695</td></tr><tr><td align="left" id="jir12217-ent-0099">19. Cannot find clothes, shoes, toys or books even with instructions</td><td id="jir12217-ent-0100" align="left">.772</td><td id="jir12217-ent-0101" /><td id="jir12217-ent-0102" /></tr><tr><td align="left" id="jir12217-ent-0103">20. Takes a long time to feel comfortable in new places/situations</td><td align="left" id="jir12217-ent-0104" /><td align="char" char="." id="jir12217-ent-0105">.495</td><td id="jir12217-ent-0106" /></tr><tr><td align="left" id="jir12217-ent-0107">21. Mood changes frequently</td><td align="left" id="jir12217-ent-0108" /><td align="char" char="." id="jir12217-ent-0109">.662</td><td id="jir12217-ent-0110" /></tr><tr><td align="left" id="jir12217-ent-0111">22. Makes silly mistakes on things he/she can do</td><td id="jir12217-ent-0112" align="left">—</td><td id="jir12217-ent-0113" align="left">—</td><td id="jir12217-ent-0114" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0115">23. Is fidgety, restless or squirmy</td><td align="left" id="jir12217-ent-0116" /><td id="jir12217-ent-0117" /><td align="char" char="." id="jir12217-ent-0118">.418</td></tr><tr><td align="left" id="jir12217-ent-0119">24. Has trouble following established routines for sleep, eating or play</td><td id="jir12217-ent-0120" align="left">—</td><td id="jir12217-ent-0121" align="left">—</td><td id="jir12217-ent-0122" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0123">25. Is bothered by loud noises, bright lights or certain smells</td><td id="jir12217-ent-0124" align="left">—</td><td id="jir12217-ent-0125" align="left">—</td><td id="jir12217-ent-0126" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0127">26. Small events trigger big reactions</td><td align="left" id="jir12217-ent-0128" /><td align="char" char="." id="jir12217-ent-0129">.759</td><td id="jir12217-ent-0130" /></tr><tr><td align="left" id="jir12217-ent-0131">27. Has trouble with activities or tasks that have more than one step</td><td id="jir12217-ent-0132" align="left">.689</td><td id="jir12217-ent-0133" /><td id="jir12217-ent-0134" /></tr><tr><td align="left" id="jir12217-ent-0135">28. Is impulsive</td><td align="left" id="jir12217-ent-0136" /><td id="jir12217-ent-0137" /><td align="char" char="." id="jir12217-ent-0138">.692</td></tr><tr><td align="left" id="jir12217-ent-0139">29. Has trouble thinking of different way to solve problems when stuck</td><td id="jir12217-ent-0140" align="left">.600</td><td id="jir12217-ent-0141" /><td id="jir12217-ent-0142" /></tr><tr><td align="left" id="jir12217-ent-0143">30. Is disturbed by changes in the environment</td><td align="left" id="jir12217-ent-0144" /><td align="char" char="." id="jir12217-ent-0145">.503</td><td id="jir12217-ent-0146" /></tr><tr><td align="left" id="jir12217-ent-0147">31. Angry/tearful outbursts are intense but end suddenly</td><td align="left" id="jir12217-ent-0148" /><td align="char" char="." id="jir12217-ent-0149">.508</td><td id="jir12217-ent-0150" /></tr><tr><td align="left" id="jir12217-ent-0151">32. Needs help from adult to stay on task</td><td id="jir12217-ent-0152" align="left">.508</td><td id="jir12217-ent-0153" /><td id="jir12217-ent-0154" /></tr><tr><td align="left" id="jir12217-ent-0155">33. Does not notice when his/her behaviour causes negative reactions</td><td id="jir12217-ent-0156" align="left">.431</td><td id="jir12217-ent-0157" /><td id="jir12217-ent-0158" /></tr><tr><td align="left" id="jir12217-ent-0159">34. Leaves messes that others have to clean up even after instruction</td><td id="jir12217-ent-0160" align="left">.598</td><td id="jir12217-ent-0161" /><td id="jir12217-ent-0162" /></tr><tr><td align="left" id="jir12217-ent-0163">35. Has trouble changing activities</td><td id="jir12217-ent-0164" align="left">—</td><td id="jir12217-ent-0165" align="left">—</td><td id="jir12217-ent-0166" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0167">36. Reacts more strongly to situations than other children</td><td align="left" id="jir12217-ent-0168" /><td align="char" char="." id="jir12217-ent-0169">.586</td><td id="jir12217-ent-0170" /></tr><tr><td align="left" id="jir12217-ent-0171">37. Forgets what he/she is doing in the middle of an activity</td><td id="jir12217-ent-0172" align="left">.674</td><td id="jir12217-ent-0173" /><td id="jir12217-ent-0174" /></tr><tr><td align="left" id="jir12217-ent-0175">38. Does not realise that certain actions bother others</td><td id="jir12217-ent-0176" align="left">.503</td><td id="jir12217-ent-0177" /><td id="jir12217-ent-0178" /></tr><tr><td align="left" id="jir12217-ent-0179">39. Gets caught up in the small details of task and misses the main idea</td><td id="jir12217-ent-0180" align="left">.436</td><td id="jir12217-ent-0181" /><td id="jir12217-ent-0182" /></tr><tr><td align="left" id="jir12217-ent-0183">40. Has trouble ‘joining in’ at unfamiliar social events</td><td id="jir12217-ent-0184" align="left">.470</td><td id="jir12217-ent-0185" /><td id="jir12217-ent-0186" /></tr><tr><td align="left" id="jir12217-ent-0187">41. Is easily overwhelmed or overstimulated by typical daily activities</td><td id="jir12217-ent-0188" align="left">—</td><td id="jir12217-ent-0189" align="left">—</td><td id="jir12217-ent-0190" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0191">42. Has trouble finishing tasks</td><td id="jir12217-ent-0192" align="left">.635</td><td id="jir12217-ent-0193" /><td id="jir12217-ent-0194" /></tr><tr><td align="left" id="jir12217-ent-0195">43. Gets out of control more than playmates</td><td align="left" id="jir12217-ent-0196" /><td id="jir12217-ent-0197" /><td align="char" char="." id="jir12217-ent-0198">.791</td></tr><tr><td align="left" id="jir12217-ent-0199">44. Cannot find things in room even when given specific instructions</td><td id="jir12217-ent-0200" align="left">.727</td><td id="jir12217-ent-0201" /><td id="jir12217-ent-0202" /></tr><tr><td align="left" id="jir12217-ent-0203">45. Resists change of routine, foods, places, etc.</td><td align="left" id="jir12217-ent-0204" /><td align="char" char="." id="jir12217-ent-0205">.543</td><td id="jir12217-ent-0206" /></tr><tr><td align="left" id="jir12217-ent-0207">46. After having a problem, will stay disappointed for a long time</td><td align="left" id="jir12217-ent-0208" /><td align="char" char="." id="jir12217-ent-0209">.419</td><td id="jir12217-ent-0210" /></tr><tr><td align="left" id="jir12217-ent-0211">47. Cannot stay on the same topic when talking</td><td id="jir12217-ent-0212" align="left">—</td><td id="jir12217-ent-0213" align="left">—</td><td id="jir12217-ent-0214" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0215">48. Talks or plays too loudly</td><td id="jir12217-ent-0216" align="left">—</td><td id="jir12217-ent-0217" align="left">—</td><td id="jir12217-ent-0218" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0219">49. Does not complete tasks even after given directions</td><td id="jir12217-ent-0220" align="left">.681</td><td id="jir12217-ent-0221" /><td id="jir12217-ent-0222" /></tr><tr><td align="left" id="jir12217-ent-0223">50. Acts overwhelmed or overstimulated in crowded, busy situations</td><td id="jir12217-ent-0224" align="left">—</td><td id="jir12217-ent-0225" align="left">—</td><td id="jir12217-ent-0226" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0227">51. Has trouble getting started on activities or tasks even after instructed</td><td id="jir12217-ent-0228" align="left">.709</td><td id="jir12217-ent-0229" /><td id="jir12217-ent-0230" /></tr><tr><td align="left" id="jir12217-ent-0231">52. Acts too wild or out of control</td><td align="left" id="jir12217-ent-0232" /><td id="jir12217-ent-0233" /><td align="char" char="." id="jir12217-ent-0234">.790</td></tr><tr><td align="left" id="jir12217-ent-0235">53. Does not try as hard as his/her ability on activities</td><td id="jir12217-ent-0236" align="left">—</td><td id="jir12217-ent-0237" align="left">—</td><td id="jir12217-ent-0238" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0239">54. Has trouble putting the brakes on actions even after being asked</td><td align="left" id="jir12217-ent-0240" /><td id="jir12217-ent-0241" /><td align="char" char="." id="jir12217-ent-0242">.599</td></tr><tr><td align="left" id="jir12217-ent-0243">55. Unable to finish describing an event, person or story</td><td id="jir12217-ent-0244" align="left">—</td><td id="jir12217-ent-0245" align="left">—</td><td id="jir12217-ent-0246" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0247">56. Completes tasks or activities too quickly</td><td id="jir12217-ent-0248" align="left">—</td><td id="jir12217-ent-0249" align="left">—</td><td id="jir12217-ent-0250" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0251">57. Is unaware when he/she does well and not well</td><td id="jir12217-ent-0252" align="left">.560</td><td id="jir12217-ent-0253" /><td id="jir12217-ent-0254" /></tr><tr><td align="left" id="jir12217-ent-0255">58. Gets easily sidetracked during activities</td><td id="jir12217-ent-0256" align="left">.593</td><td id="jir12217-ent-0257" /><td id="jir12217-ent-0258" /></tr><tr><td align="left" id="jir12217-ent-0259">59. Has trouble remembering, even after a brief period of time</td><td id="jir12217-ent-0260" align="left">.630</td><td id="jir12217-ent-0261" /><td id="jir12217-ent-0262" /></tr><tr><td align="left" id="jir12217-ent-0263">60. Becomes too silly</td><td id="jir12217-ent-0264" align="left">—</td><td id="jir12217-ent-0265" align="left">—</td><td id="jir12217-ent-0266" align="left">—</td></tr><tr><td align="left" id="jir12217-ent-0267">61. Has a short attention span</td><td id="jir12217-ent-0268" align="left">.444</td><td id="jir12217-ent-0269" /><td id="jir12217-ent-0270" /></tr><tr><td align="left" id="jir12217-ent-0271">62. Plays recklessly in situations where he/she could be hurt</td><td align="left" id="jir12217-ent-0272" /><td id="jir12217-ent-0273" /><td align="char" char="." id="jir12217-ent-0274">.409</td></tr><tr><td align="left" id="jir12217-ent-0275">63. Is unaware when he/she performs a task right or wrong</td><td id="jir12217-ent-0276" align="left">.698</td><td id="jir12217-ent-0277" /><td id="jir12217-ent-0278" /></tr></table> </ephtml> </p> <p>Cronbach's alpha was calculated as a measure of the internal consistency of each factor. All alpha coefficients fell within an acceptable range:.94 for the 26‐item Emergent Metacognition factor,.90 for the 15‐item Flexibility factor and.87 for the 7‐item Inhibition factor.</p> <hd id="AN0111334429-13">Comparisons of executive functions between clinically derived groups</hd> <p>Multivariate Analysis of Variance (MANOVA) was performed using clinical group membership as the independent variable and the three BRIEF‐P factors constructed from the aforementioned factor analysis as dependent variables. A significant multivariate effect was found [F(<reflink idref="bib2" id="ref45">2</reflink>, 177) = 15.54, P < .001]. All three of the subsequent univariate tests were statistically significant (see Table [NaN] and Fig. [NaN] ). Bonferroni corrected post hoc tests were used to identify significant differences between individual clinical groups, as reported below and in Table [NaN] .</p> <p>MANOVA of clinical group membership on BRIEF‐P factors: univariate effects</p> <p> <ephtml> <table><tr><th id="jir12217-ent-0279" /><th align="left" id="jir12217-ent-0280">F value</th><th align="left" id="jir12217-ent-0281">Df between, within</th><th align="left" id="jir12217-ent-0282">P value</th><th align="left" id="jir12217-ent-0283">Eta squared</th></tr><tr><td id="jir12217-ent-0284" align="left">BRIEF‐P: Emergent Metacognition</td><td id="jir12217-ent-0285" align="left">23.44</td><td id="jir12217-ent-0286" align="left">2, 178</td><td id="jir12217-ent-0287" align="left">.000</td><td id="jir12217-ent-0288" align="left">.208</td></tr><tr><td id="jir12217-ent-0289" align="left">BRIEF‐P: Flexibility</td><td id="jir12217-ent-0290" align="left">11.95</td><td id="jir12217-ent-0291" align="left">2, 180</td><td id="jir12217-ent-0292" align="left">.000</td><td id="jir12217-ent-0293" align="left">.117</td></tr><tr><td id="jir12217-ent-0311" align="left">BRIEF‐P: Inhibition</td><td id="jir12217-ent-0312" align="left">25.85</td><td id="jir12217-ent-0313" align="left">2, 179</td><td id="jir12217-ent-0314" align="left">.000</td><td id="jir12217-ent-0315" align="left">.224</td></tr></table> </ephtml> </p> <p>Clinical group comparisons on BRIEF‐P factors</p> <p> <ephtml> <table><tr><th id="jir12217-ent-0294" align="left">Subscale</th><th align="left" id="jir12217-ent-0295">Typical DS (n = 24)</th><th align="left" id="jir12217-ent-0296">DS + ASD (n = 67)</th><th align="left" id="jir12217-ent-0297">DS + DBD (n = 98)</th></tr><tr><td id="jir12217-ent-0298" align="left">Mean (SD)</td></tr><tr><td id="jir12217-ent-0299" align="left">Emergent metacognition</td><td id="jir12217-ent-0300" align="left">1.67 (0.42)</td><td id="jir12217-ent-0301" align="left">2.34 (0.47)</td><td align="left" id="jir12217-ent-0302">2.14 (0.35)</td></tr><tr><td id="jir12217-ent-0303" align="left">Flexibility</td><td id="jir12217-ent-0304" align="left">1.40 (0.22)</td><td id="jir12217-ent-0305" align="left">1.88 (0.48)</td><td id="jir12217-ent-0306" align="left">1.86 (0.45)</td></tr><tr><td id="jir12217-ent-0307" align="left">Inhibition</td><td id="jir12217-ent-0308" align="left">1.41 (0.32)</td><td id="jir12217-ent-0309" align="left">2.00 (0.57)</td><td id="jir12217-ent-0310" align="left">2.27 (0.55)</td></tr></table> </ephtml> </p> <ulist> <item>2 †Indicates significant differences (P < .05) between typical DS and DS + ASD and between typical DS and DS + DBD</item> <item>3 ‡Indicates a significant difference (P < .05) between DS + ASD and DS + DBD</item> </ulist> <p>On the Emergent Metacognition factor, the Typical DS group evidenced significantly lower scores (fewer problems) than either the DS + ASD group or the DS + DBD group. The DS + ASD group demonstrated higher scores than did the DS + DBD group.</p> <p>On the Flexibility factor, the Typical DS group evidenced significantly lower scores (more flexibility) than either the DS + ASD group or the DS + DBD group. Significant differences were not found in flexibility between the DS + ASD and the DS + DBD groups.</p> <p>On the Inhibition factor, the Typical DS group evidenced significantly lower scores (better inhibition) than either the DS + ASD or the DS + DBD group. The DS + DBD group demonstrated higher scores than did the DS + ASD group.</p> <hd id="AN0111334429-14">Relationship between intellectual and executive functions</hd> <p>Using independent samples t‐tests, we compared the cognitively lower‐functioning group with the cognitively higher‐functioning group on each of the three BRIEF‐P factors. No significant group differences were found on any factor (Emergent Metacognition t(<reflink idref="bib76" id="ref46">76</reflink>) = −0.719, P = .475; Flexibility t(<reflink idref="bib78" id="ref47">78</reflink>) = −0.101, P = .920; Inhibition t(<reflink idref="bib77" id="ref48">77</reflink>) = −0.054, P = .957). Among the subsample of participants for whom norm‐referenced estimates of cognitive functioning were available (n = 59), the correlations between cognitive functioning and the BRIEF‐P Flexibility and Inhibition were not statistically significant (r = −.20, P = .145 and r = −.03, P = .84, respectively); however, there was a significant negative correlation between IQ and the BRIEF‐P Emergent Metacognition factor (r = −.28, P = .04).</p> <hd id="AN0111334429-15">Discussion</hd> <p>The first goal of the present study was to evaluate the psychometric properties of the BRIEF‐P when used out of age range for clinic‐referred youth with DS. The results of our exploratory factory analysis suggest that a three‐factor solution best fits the data for our clinic‐based sample of youth with DS, and that these three factors largely correspond, in theme, to the three theoretically driven composite indices originally proposed for use with the measure among pre‐schoolers, and to other behavioural models of EF (Barkley [<reflink idref="bib3" id="ref49">3</reflink>] ; Gioia et al. [<reflink idref="bib22" id="ref50">22</reflink>] ). The theoretically derived Emergent Metacognition index is comprised of the Working Memory and Plan/Organise subscales, with all of the items from each subscale contributing to the total index score. Our empirically derived Emergent Metacognition factor is comprised of items from the Working Memory (50% of the items on this factor), Plan/Organise (35%), Inhibit (12%) and Shift (4%) subscales. Similarly, the theoretically derived Flexibility index is comprised of all items from the Shift and Emotional Control subscales. Our empirically derived Flexibility factor is made up of items from the same subscales, with 40% of items on this factor taken from the Shift subscale and 60% from the Emotional Control subscale. Finally, the theoretically derived Inhibitory Self Control index is comprised of all items from the Inhibit and Emotional Control subscales; however, our empirically derived Inhibitory Self Control factor is made up entirely of items from the Inhibit subscale. So, among clinic‐referred youth with DS, our findings suggest that the BRIEF may produce a more focused Inhibition factor, a fairly similar Flexibility factor, and a similar, though not as clean, Emergent Metacognition factor, compared to the original BRIEF‐P index conceptualisations. Taken together, these findings offer preliminary psychometric evidence to support the idea that the BRIEF‐P is capturing similar constructs in clinic‐referred youth with DS as it does in pre‐schoolers, although we cannot generalise these findings to the full population of youth with DS, because of the behavioural and psychiatric differences that result from the clinic‐based nature of the present sample.</p> <p>In addition, these findings offer preliminary evidence of the BREIF‐P factors' utility in providing empirical support for behaviourally based psychiatric diagnoses for which youth with DS are at risk. While clinic‐referred youth with DS who do not have additional psychiatric diagnoses evidence relatively few problems across all executive functions, youth with DS + ASD are likely to show the greatest difficulty in Emergent Metacognition, with more moderate difficulties in Flexibility and Inhibition. In comparison, youth with DS + DBD are likely to show considerable difficulties in both the Emergent Metacognition and Inhibition domains, with more moderate problems with Flexibility.</p> <p>Because there is often a question as to whether functional behavioural differences are associated with general intellectual deficits, a preliminary evaluation of the relationship between intellectual ability and executive functions among our sample of clinic‐referred youth was included. When dichotomised into a lower functioning group and a somewhat higher functioning group, no group differences were found on executive functions; however, within the higher functioning norm‐referenced IQ group, children with lower IQs were rated more poorly on the Emergent Metacognition factor. This significant association stands to reason, given the cognitive nature of many of the behaviours that comprise this factor (e.g. having trouble with multi‐step tasks, remembering things, etc.). Given that intellectual functioning was identified as being significantly lower among the DS + ASD group, it is possible that this group's relatively greater difficulties in the area of Emergent Cognition are, at least in part, a result of their lower level of cognitive ability. Taken together, these findings offer preliminary support for broad use of the BRIEF‐P with clinic‐referred youth with DS, with the caveat that an individual's intellectual ability may be significantly related to their Emergent Cognition score in this population.</p> <hd id="AN0111334429-16">Clinical implications</hd> <p>The findings of this study have important clinical implications for families, educators and clinicians, as they try to understand the underpinnings of specific behaviours, particularly as they impact day to day functioning, observed within children diagnosed with DS. First, this study demonstrates that EFs, which play an important role in the achievement of various adaptive functions (e.g. (Gilotty et al. [<reflink idref="bib20" id="ref51">20</reflink>] )), can be assessed in a meaningful way via parent behaviour ratings for clinic‐referred youth with DS. If clinicians and educators can develop a better understanding of the root of a given child's adaptive or behavioural difficulties via these ratings, then this would allow for more appropriate supports to be put in place to better enhance adaptive skill acquisition among these youth.</p> <p>Second, in many cases, some level of EF dysfunction and associated maladaptive behaviour may be present among youth with DS prior to reaching threshold criteria for a DSM‐based disorder in childhood or adolescence. Early identification of specific impairment in EF can facilitate caretaker education and intervention for those children most at risk for being diagnosed with a serious neurobehavioural disorder.</p> <hd id="AN0111334429-17">Limitations</hd> <p>The conclusions drawn by this study are subject to a few limitations. First, the study sample is derived from a clinical cohort with a referral bias towards individuals with high levels of maladaptive behaviour, most of whom (87%) met criteria for a DSM‐IV‐TR disorder. While this may limit the ability to generalise our findings to the larger population of children with DS, the inclusion of a typically behaving DS comparison group ascertained through the same clinic provides a basis for useful comparison. Second, although equal in representation across all DSM‐IV‐TR groups, information on cognitive function was only available for 45% of our study sample. Inconsistency in test instruments, personnel and procedures introduces methodological variance which cannot be controlled for in this type of clinical sample. Thus the cognitive findings, although generally consistent with clinical expectation, should be considered preliminary at this time. Third, DSM‐IV‐TR assignment was made by a single un‐blinded clinician, and only a primary diagnosis was given; thus secondary diagnoses and comorbid features remain unconsidered. Fourth, although DSM‐IV‐TR represents a consensus‐derived diagnostic system, it lacks the rigour to consistently distinguish deficits in social communication and reciprocity from other types of impairment in sociability in children with severe ID, a weakness shared by observational schedules commonly used to diagnose autism spectrum disorders ((Starr et al. [<reflink idref="bib41" id="ref52">41</reflink>] ), (DiGuiseppi et al. [<reflink idref="bib13" id="ref53">13</reflink>] )). In future studies, a more rigorous approach to diagnostic classification (e.g. semi‐structured psychiatric interview by two independent and blinded clinicians) should be used to increase the reliability of clinical diagnoses and delineate comorbid anxious, obsessive–compulsive, mood or other features associated with particular clinical diagnoses in this population.</p> <hd id="AN0111334429-18">Future directions</hd> <p>This study provides psychometric evidence to support the use of the BRIEF‐P with clinic‐referred children with Down Syndrome and suggests that the measure may have utility in describing the unique executive and behavioural strengths and weaknesses of children with Down Syndrome complicated by an psychiatric condition. Further study of the emergence and developmental trajectory of specific types of EF impairment in young children with DS is necessary to elucidate whether changes in the behavioural manifestations of EF occur with development. Future research will also need to investigate how common concerns among children with DS (e.g. communication difficulties, sleep disturbances; (Breslin et al. [<reflink idref="bib4" id="ref54">4</reflink>] )) impact EF development. It will also be important to directly examine the relationship between EF and adaptive skills in this population, particularly as executive skills may relate to a child's ability to self‐regulate and develop greater independence. One of the major challenges in evaluating the role of intellectual abilities in executive functions is the lack of adequate standardised instruments for the measurement of intellectual abilities in youth with DS. Future research, and clinical practice, would benefit substantially from the development and utilisation of standardised measures of intelligence that are appropriate for youth with DS.</p> <ref id="AN0111334429-19"> <title>References</title> <blist> <bibl id="bib1" idref="ref40" type="bt">1</bibl> <bibtext>American Psychiatric Association. ( 2000 ) Diagnostic and Statistical Manual of Mental Disorders: DSM‐IV‐TR®. American Psychiatric Pub, Arlington, VA. </bibtext> </blist> <blist> <bibl id="bib2" idref="ref14" type="bt">2</bibl> <bibtext>Baddeley, A. & Jarrold C. ( 2007 ) Working memory and Down syndrome. Journal of Intellectual Disability Research 51, 925 – 31. </bibtext> </blist> <blist> <bibl id="bib3" idref="ref49" type="bt">3</bibl> <bibtext>Barkley R. A. ( 1997 ) Behavioral inhibition, sustained attention, and executive functions: constructing a unifying theory of ADHD. 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( 1999 ) Social competence in children with autism, Down syndrome and other developmental delays: a longitudinal study. Monographs of the Society for Research in Child Development, Serial no.256, 64, 109 – 13. </bibtext> </blist> <blist> <bibl id="bib40" idref="ref2" type="bt">40</bibl> <bibtext>Silverman W. ( 2007 ) Down syndrome: cognitive phenotype. Mental Retardation and Developmental Disabilities Research Reviews 13, 228 – 36. </bibtext> </blist> <blist> <bibl id="bib41" idref="ref52" type="bt">41</bibl> <bibtext>Starr E. M., Berument S. K., Tomlins M., Papanikolaou K. & Rutter M. ( 2005 ) Brief report: autism in individuals with Down syndrome. Journal of Autism and Developmental Disorders 35, 665 – 73. </bibtext> </blist> <blist> <bibl id="bib42" idref="ref16" type="bt">42</bibl> <bibtext>Toplak M. E., West R. F. & Stanovich K. E. ( 2013 ) Practitioner review: do performance‐based measures and ratings of executive function assess the same construct? Journal of Child Psychology and Psychiatry 54, 131 – 43. </bibtext> </blist> <blist> <bibl id="bib43" idref="ref22" type="bt">43</bibl> <bibtext>Van Duijn G., Dijkxhoorn Y., Scholte E. & Berckelaer‐Onnes V. ( 2010 ) The development of adaptive skills in young people with Down syndrome. Journal of Intellectual Disability Research 54, 943 – 54. </bibtext> </blist> </ref> <p>Graph: Profile of BRIEF‐P factor scores across clinically derived groups. BRIEF‐P factors, as identified by exploratory factor analysis, can be used to differentiate typical youth with DS from youth with DS and co‐occurring Autism Spectrum Disorder, and from youth with DS and co‐occurring Disruptive Behaviour Disorder. The DS and ASD group is distinguished by particularly large deficits in Emergent Metacognition, while the DS and DBD group demonstrates significant deficits in both Emergent Metacognition and Inhibition.</p> <aug> <p>By A. E. Pritchard; S. Kalback; M. McCurdy and G. T. Capone</p> </aug> <nolink nlid="nl1" bibid="bib79" firstref="ref43"></nolink> <nolink nlid="nl2" bibid="bib76" firstref="ref46"></nolink> <nolink nlid="nl3" bibid="bib78" firstref="ref47"></nolink> <nolink nlid="nl4" bibid="bib77" firstref="ref48"></nolink>
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  Data: Executive Functions among Youth with Down Syndrome and Co-Existing Neurobehavioural Disorders
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  Data: <searchLink fieldCode="AR" term="%22Pritchard%2C+A%2E+E%2E%22">Pritchard, A. E.</searchLink><br /><searchLink fieldCode="AR" term="%22Kalback%2C+S%2E%22">Kalback, S.</searchLink><br /><searchLink fieldCode="AR" term="%22McCurdy%2C+M%2E%22">McCurdy, M.</searchLink>
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  Data: <searchLink fieldCode="SO" term="%22Journal+of+Intellectual+Disability+Research%22"><i>Journal of Intellectual Disability Research</i></searchLink>. Dec 2015 59(12):1130-1141.
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  Data: Wiley-Blackwell. 350 Main Street, Malden, MA 02148. Tel: 800-835-6770; Tel: 781-388-8598; Fax: 781-388-8232; e-mail: cs-journals@wiley.com; Web site: http://www.wiley.com/WileyCDA
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  Data: 12
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  Data: Journal Articles<br />Reports - Research
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  Data: <searchLink fieldCode="DE" term="%22Executive+Function%22">Executive Function</searchLink><br /><searchLink fieldCode="DE" term="%22Down+Syndrome%22">Down Syndrome</searchLink><br /><searchLink fieldCode="DE" term="%22Neurological+Impairments%22">Neurological Impairments</searchLink><br /><searchLink fieldCode="DE" term="%22Behavior+Problems%22">Behavior Problems</searchLink><br /><searchLink fieldCode="DE" term="%22Comorbidity%22">Comorbidity</searchLink><br /><searchLink fieldCode="DE" term="%22Behavior+Rating+Scales%22">Behavior Rating Scales</searchLink><br /><searchLink fieldCode="DE" term="%22Psychometrics%22">Psychometrics</searchLink><br /><searchLink fieldCode="DE" term="%22Autism%22">Autism</searchLink><br /><searchLink fieldCode="DE" term="%22Pervasive+Developmental+Disorders%22">Pervasive Developmental Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Factor+Analysis%22">Factor Analysis</searchLink><br /><searchLink fieldCode="DE" term="%22Metacognition%22">Metacognition</searchLink><br /><searchLink fieldCode="DE" term="%22Inhibition%22">Inhibition</searchLink><br /><searchLink fieldCode="DE" term="%22Intellectual+Disability%22">Intellectual Disability</searchLink>
– Name: DOI
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  Group: ID
  Data: 10.1111/jir.12217
– Name: ISSN
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  Group: ISSN
  Data: 0964-2633
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Background: Executive function (EF) deficits are :a recognised component of the cognitive phenotype of youth with Down Syndrome (DS). Recent research in this area emphasises the use of behaviour ratings, such as the Behavior Rating Inventory of Executive Functions-Preschool Version (BRIEF-P), to capture the real-world applications of executive functions. To account for the intellectual functioning of youth with DS, this measure is used out of age range; however, its psychometric properties when used in this fashion are unknown. The goals of this study are to evaluate psychometric characteristics of the BRIEF-P among youth with DS and to examine the pattern of EF strengths/weaknesses in children with DS and co-occurring psychiatric conditions. Method: A total of 188 clinically referred youth with DS, ages 3-13 were rated by their caregivers using the BRIEF-P. These youth were evaluated by a clinician with expertise in DS and were characterised as having no co-occurring behavioural disorder (Typical DS group), co-occurring Autism Spectrum Disorder (DS?+?ASD) or co-occurring Disruptive Behaviour Disorder (DS?+?DBD). Results: An exploratory factor analysis of item-level BRIEF-P data from clinically referred youth with DS supported the theoretically derived three-factor structure originally proposed for the BRIEF-P (Emergent Metacognition, Flexibility and Inhibitory Self-Control); however, the item composition of each factor varied somewhat in comparison to the original structure of the measure. Group comparisons indicate that, while youth with typical DS evidence fewer executive function difficulties across all domains, youth with DS?+?ASD show the greatest weaknesses in Emergent Metacognition, and youth with DS?+?DBD show significant difficulties in both Emergent Metacognition and Inhibition. Conclusions: These findings offer preliminary support for use of the BRIEF-P with clinically referred youth with Down Syndrome. Some scoring modifications may be necessary if the theoretically derived index scores are to be used with this population. BRIEF-P scores may offer an empirical basis for differentiating DS youth with varying behavioural profiles.
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  Data: 2016
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  Data: EJ1087751
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        Value: 10.1111/jir.12217
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      – Text: English
    PhysicalDescription:
      Pagination:
        PageCount: 12
        StartPage: 1130
    Subjects:
      – SubjectFull: Executive Function
        Type: general
      – SubjectFull: Down Syndrome
        Type: general
      – SubjectFull: Neurological Impairments
        Type: general
      – SubjectFull: Behavior Problems
        Type: general
      – SubjectFull: Comorbidity
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      – SubjectFull: Behavior Rating Scales
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      – SubjectFull: Psychometrics
        Type: general
      – SubjectFull: Autism
        Type: general
      – SubjectFull: Pervasive Developmental Disorders
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      – SubjectFull: Factor Analysis
        Type: general
      – SubjectFull: Metacognition
        Type: general
      – SubjectFull: Inhibition
        Type: general
      – SubjectFull: Intellectual Disability
        Type: general
    Titles:
      – TitleFull: Executive Functions among Youth with Down Syndrome and Co-Existing Neurobehavioural Disorders
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          Name:
            NameFull: Pritchard, A. E.
      – PersonEntity:
          Name:
            NameFull: Kalback, S.
      – PersonEntity:
          Name:
            NameFull: McCurdy, M.
    IsPartOfRelationships:
      – BibEntity:
          Dates:
            – D: 01
              M: 12
              Type: published
              Y: 2015
          Identifiers:
            – Type: issn-print
              Value: 0964-2633
          Numbering:
            – Type: volume
              Value: 59
            – Type: issue
              Value: 12
          Titles:
            – TitleFull: Journal of Intellectual Disability Research
              Type: main
ResultId 1