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Language Comprehension in Boys with Fragile X Syndrome and Boys with Down Syndrome

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Title: Language Comprehension in Boys with Fragile X Syndrome and Boys with Down Syndrome
Language: English
Authors: Price, J., Roberts, J., Vandergrift, N., Martin, G.
Source: Journal of Intellectual Disability Research. Apr 2007 51(4):318-326.
Availability: Blackwell Publishing. 350 Main Street, Malden, MA 02148. Tel: 800-835-6770; Tel: 781-388-8599; Fax: 781-388-8232; e-mail: customerservices@blackwellpublishing.com; Web site: http://www.blackwellpublishing.com/jnl_default.asp
Peer Reviewed: Y
Page Count: 9
Publication Date: 2007
Document Type: Journal Articles
Reports - Research
Descriptors: Morphology (Languages), Down Syndrome, Syntax, Sentences, Receptive Language, Males, Listening Comprehension, Language Skills, Autism, Genetics, Congenital Impairments, Mental Retardation, Grammar, Pervasive Developmental Disorders, Comparative Analysis, Vocabulary
Assessment and Survey Identifiers: Test for Auditory Comprehension of Language
DOI: 10.1111/j.1365-2788.2006.00881.x
ISSN: 0964-2633
Abstract: Background: Fragile X syndrome (FXS) is the most common known inherited cause of intellectual disability, yet very few studies have explored the language comprehension skills of children with FXS. We examined the receptive vocabulary, grammatical morphology and syntax skills of boys with FXS (who were additionally classified as having autism, autism spectrum, or no autism) and compared them to boys with Down syndrome (DS) and typically developing (TD) boys at similar non-verbal developmental levels. Methods: The Vocabulary, Grammatical Morphology, and Elaborated Phrases and Sentences subtests of the Test for Auditory Comprehension of Language--3rd Edition (TACL-3) were administered annually up to three times to assess the language comprehension skills of 35 boys with FXS without autism, 24 boys with FXS with autism spectrum, 19 boys with FXS with autism, 45 boys with DS and 40 TD boys at similar non-verbal cognitive levels. Results: After controlling for non-verbal cognition and maternal education levels, we found that the three groups of boys with FXS did not differ from each other but scored lower than the TD boys in language comprehension. The boys with DS scored lower in language comprehension than boys with FXS without autism and TD boys. For all of the groups, scores for receptive vocabulary, grammatical morphology and syntax did not differ. Conclusions: Boys with FXS and boys with DS differed in receptive language levels, demonstrating unique language profiles for each syndrome. Language comprehension appears to be an important area to target in assessment and intervention for both populations.
Abstractor: Author
Number of References: 51
Entry Date: 2007
Accession Number: EJ754595
Database: ERIC
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  Value: <anid>AN0024165157;eul01apr.07;2019Jun04.10:25;v2.2.500</anid> <title id="AN0024165157-1">Language comprehension in boys with fragile X syndrome and boys with Down syndrome. </title> <p>Background  Fragile X syndrome (FXS) is the most common known inherited cause of intellectual disability, yet very few studies have explored the language comprehension skills of children with FXS. We examined the receptive vocabulary, grammatical morphology and syntax skills of boys with FXS (who were additionally classified as having autism, autism spectrum, or no autism) and compared them to boys with Down syndrome (DS) and typically developing (TD) boys at similar non‐verbal developmental levels. Methods  The Vocabulary, Grammatical Morphology, and Elaborated Phrases and Sentences subtests of the Test for Auditory Comprehension of Language – 3rd Edition (TACL‐3) were administered annually up to three times to assess the language comprehension skills of 35 boys with FXS without autism, 24 boys with FXS with autism spectrum, 19 boys with FXS with autism, 45 boys with DS and 40 TD boys at similar non‐verbal cognitive levels. Results  After controlling for non‐verbal cognition and maternal education levels, we found that the three groups of boys with FXS did not differ from each other but scored lower than the TD boys in language comprehension. The boys with DS scored lower in language comprehension than boys with FXS without autism and TD boys. For all of the groups, scores for receptive vocabulary, grammatical morphology and syntax did not differ. Conclusions  Boys with FXS and boys with DS differed in receptive language levels, demonstrating unique language profiles for each syndrome. Language comprehension appears to be an important area to target in assessment and intervention for both populations.</p> <p>Keywords: fragile X syndrome; language comprehension; neurodevelopmental disorders; Down syndrome</p> <p>Fragile X syndrome (FXS) is the most common known inherited cause of intellectual disability (ID) ([<reflink idref="bib28" id="ref1">28</reflink>]). Relatively few studies have examined language skills in children with FXS and even fewer have focused on language comprehension skills ([<reflink idref="bib2" id="ref2">2</reflink>]), a building block for expressive language, cognitive and academic skills. Moreover, a major goal of current developmental disabilities research is to compare language phenotypes across syndromes to determine whether each syndrome is characterized by a distinct profile, or whether language characteristics can be explained more generally by the presence of ID ([<reflink idref="bib49" id="ref3">49</reflink>]; [<reflink idref="bib43" id="ref4">43</reflink>]). Therefore, we examined the language comprehension skills of boys with FXS (who were additionally classified as having autism, autism spectrum, or no autism) and compared them to boys with Down syndrome (DS) and typically developing (TD) boys at similar non‐verbal developmental levels. We studied three aspects of children's language comprehension: (<reflink idref="bib1" id="ref5">1</reflink>) vocabulary (individual words); (<reflink idref="bib2" id="ref6">2</reflink>) grammatical morphology (prepositions and bound morphemes, such as plural <emph>s</emph>); and (<reflink idref="bib3" id="ref7">3</reflink>) syntax (multiword syntactic patterns such as active and passive voice and indirect objects).</p> <p>Fragile X syndrome occurs in one of every 4000 individuals ([<reflink idref="bib50" id="ref8">50</reflink>]; [<reflink idref="bib16" id="ref9">16</reflink>]). Inadequate amounts of the Fragile X Mental Retardation Protein (FMRP), essential for normal brain functioning, are produced ([<reflink idref="bib17" id="ref10">17</reflink>]; [<reflink idref="bib29" id="ref11">29</reflink>]). Because FXS is an X‐linked disorder, boys' development is more severely affected than that of girls ([<reflink idref="bib28" id="ref12">28</reflink>]; [<reflink idref="bib32" id="ref13">32</reflink>]; [<reflink idref="bib41" id="ref14">41</reflink>]); therefore, we focused on boys in this study. DS, a genetic disorder in which there is a third chromosome 21, occurs in one of every 920 births ([<reflink idref="bib19" id="ref15">19</reflink>]; [<reflink idref="bib10" id="ref16">10</reflink>]). Compared to individuals with FXS, individuals with DS have similar degrees of cognitive impairment yet some differences in language skills, comprising an ideal comparison sample.</p> <hd id="AN0024165157-2">Speech and language characteristics of fragile X syndrome and Down syndrome</hd> <p>Despite considerable individual differences, most boys with FXS show moderate to severe delays in communication skills and greater delays in expressive than receptive language ([<reflink idref="bib44" id="ref17">44</reflink>]; [<reflink idref="bib8" id="ref18">8</reflink>]; [<reflink idref="bib40" id="ref19">40</reflink>]). Fifteen per cent to 25% of individuals with FXS are diagnosed with autism ([<reflink idref="bib27" id="ref20">27</reflink>]). Although communication deficits are a defining feature of autism ([<reflink idref="bib4" id="ref21">4</reflink>]), only a few studies have investigated the relationship between autism and language skills in children with FXS. Findings suggest that children with FXS with autism have lower language skills than children with FXS without autism ([<reflink idref="bib5" id="ref22">5</reflink>], [<reflink idref="bib6" id="ref23">6</reflink>]; [<reflink idref="bib46" id="ref24">46</reflink>]; [<reflink idref="bib40" id="ref25">40</reflink>]), demonstrating the importance of examining autism status in children with FXS.</p> <p>For children with DS, language skills are more severely affected than non‐verbal cognition ([<reflink idref="bib36" id="ref26">36</reflink>]; [<reflink idref="bib26" id="ref27">26</reflink>]; [<reflink idref="bib3" id="ref28">3</reflink>]; [<reflink idref="bib51" id="ref29">51</reflink>]). Expressive language skills are poorer than receptive language skills ([<reflink idref="bib13" id="ref30">13</reflink>]; [<reflink idref="bib3" id="ref31">3</reflink>]), and syntax tends to be considerably delayed ([<reflink idref="bib13" id="ref32">13</reflink>]; [<reflink idref="bib23" id="ref33">23</reflink>]; [<reflink idref="bib3" id="ref34">3</reflink>]; [<reflink idref="bib31" id="ref35">31</reflink>]).</p> <hd id="AN0024165157-3">Language comprehension in fragile X syndrome and Down syndrome</hd> <p>The receptive vocabulary and syntax levels of adolescents and adults with FXS are similar to those of both TD mental age matches ([<reflink idref="bib3" id="ref36">3</reflink>]) and mental age matches with other forms of IDs ([<reflink idref="bib39" id="ref37">39</reflink>]). In addition, [<reflink idref="bib35" id="ref38">35</reflink>]) reported that two boys had similar levels of receptive vocabulary and non‐verbal cognition and two of three male adults had higher receptive vocabulary than non‐verbal cognition. For individuals with DS, receptive vocabulary levels appear to be similar to those of TD children of the same mental age ([<reflink idref="bib12" id="ref39">12</reflink>]; [<reflink idref="bib30" id="ref40">30</reflink>]). Some studies indicate that the syntax comprehension skills of children and adolescents with DS are commensurate with non‐verbal cognitive skills ([<reflink idref="bib12" id="ref41">12</reflink>]; [<reflink idref="bib37" id="ref42">37</reflink>]), but others have found that receptive syntax levels are lower than non‐verbal cognitive levels ([<reflink idref="bib48" id="ref43">48</reflink>]; [<reflink idref="bib30" id="ref44">30</reflink>]). [<reflink idref="bib3" id="ref45">3</reflink>]) found that the overall receptive language levels of adolescents and young adults with DS were similar to those of non‐verbal mental age matches. [<reflink idref="bib3" id="ref46">3</reflink>]) also reported that overall receptive language was lower in individuals with DS than in individuals with FXS without autism, while Roberts <emph>et al</emph>. (manuscript under review) found that receptive vocabulary was lower in boys with DS than in boys with FXS without autistic disorder.</p> <p>Given the few studies examining language comprehension in boys with FXS, the inconsistent findings regarding these skills for both individuals with FXS and individuals with DS, the current study was designed to determine whether boys with FXS (additionally classified as having autism, autism spectrum, or no autism), boys with DS and TD boys at similar non‐verbal cognitive levels showed similar patterns of receptive vocabulary, grammatical morphology and syntax acquisition.</p> <hd id="AN0024165157-4">Methods</hd> <p></p> <hd id="AN0024165157-5">Participants</hd> <p>Participants were recruited from an ongoing longitudinal study of FXS and from developmental and genetic clinics, physicians' offices, schools and childcare centres, and resided in the eastern USA. Enrollment criteria included an age of less than or equal to 16 years, an expressive vocabulary of at least 40 words, emergent word combinations, hearing threshold of 25 dB HL in the better ear across 500, 1000, 2000 and 4000 Hz, English as the primary language in the home and oral language as the primary mode of communication. Boys with DS and TD boys could not be diagnosed with autism. TD boys could not have developmental, hearing, speech or language disabilities. DNA analyses confirmed diagnoses of the full mutation in all boys with FXS.</p> <p>The School of Medicine Institutional Review Board at the University of North Carolina at Chapel Hill reviewed and approved study protocols annually. The parent or guardian provided informed consent at study entry. Additional details about recruitment and procedures have been reported previously (see [<reflink idref="bib45" id="ref47">45</reflink>]).</p> <hd id="AN0024165157-6">Boys with fragile X syndrome without autistic disorder (FXS‐Only)</hd> <p>Thirty‐five boys with FXS without autistic disorder participated. At the first assessment, the boys were between 3.9 and 15.9 years of age (M = 10.7), with a mean age equivalent score on the Brief IQ composite of the Leiter International Performance Scale – Revised (Leiter‐R; [<reflink idref="bib47" id="ref48">47</reflink>]) of 5.3 years. Eighty per cent of the boys were Caucasian, 14% were African American and 6% were other.</p> <hd id="AN0024165157-7">Boys with fragile X syndrome and autism spectrum (FXS‐Spec)</hd> <p>Twenty‐four boys with FXS with an Autism Diagnostic Observation Schedule – General (ADOS; [<reflink idref="bib33" id="ref49">33</reflink>]) score of autism spectrum participated in the study (see next section). At the first assessment, the boys were between 5.4 and 15.5 years of age (M = 9.4), and their mean age equivalent score on the Brief IQ composite of the Leiter‐R was 5.2 years. Eighty‐eight per cent were Caucasian, and 12% were African American.</p> <hd id="AN0024165157-8">Boys with fragile X syndrome and autism (FXS‐Aut)</hd> <p>Nineteen boys with FXS with an ADOS ([<reflink idref="bib33" id="ref50">33</reflink>]) score of autism participated in the study (see next section). At the first assessment, the boys were between 4.5 and 15.1 years of age (M = 10.0), and their mean age equivalent score on the Brief IQ composite of the Leiter‐R was 4.5 years. Ninety‐five per cent were Caucasian, and 5% were other.</p> <hd id="AN0024165157-9">Boys with Down syndrome (DS)</hd> <p>Forty‐five boys with DS participated. At the first assessment, the boys' ages ranged from 5.4 to 16.0 years (M = 9.6), and their mean age equivalent score on the Brief IQ composite of the Leiter‐R was 4.8 years. Eighty‐nine per cent of the boys were Caucasian, and 11% were African American.</p> <hd id="AN0024165157-10">Typically developing boys (TD)</hd> <p>Forty TD boys participated. At the first assessment, their ages ranged from 3.1 to 8.6 years (M = 5.5), and their mean age equivalent score on the Leiter‐R Brief IQ composite was 5.8 years. Seventy per cent were Caucasian, 18% African American and 12% other.</p> <hd id="AN0024165157-11">Measures</hd> <p></p> <hd id="AN0024165157-12">Language comprehension</hd> <p>The Test for Auditory Comprehension of Language – 3rd Edition (TACL‐3; [<reflink idref="bib11" id="ref51">11</reflink>]) was administered annually up to three times. The participant was asked to point to the picture (from among three choices) representing a stimulus item spoken by the examiner. The Vocabulary subtest assesses understanding of single‐word vocabulary items. The Grammatical Morphemes subtest measures comprehension of prepositions and bound morphemes. The Elaborated Phrases and Sentences subtest measures understanding of multiword syntactic patterns, such as active and passive voice and indirect and direct objects. Age equivalent scores on each subtest were calculated using published norms.</p> <hd id="AN0024165157-13">Non‐verbal cognition</hd> <p>The four subtests of the Brief IQ composite of the Leiter‐R (figure ground, form completion, sequential order and repeated patterns) were administered each year for as many as 3 years. The participant was asked to find an item in a picture, choose the next item in a sequence or arrange items in a pattern. Age equivalent scores were computed using published norms.</p> <hd id="AN0024165157-14">Autistic disorder in fragile X syndrome</hd> <p>The boys with FXS were classified as having autism, autism spectrum, or no autism using the ADOS. The ADOS is a standardized observation of children's communicative and social behaviour that discriminates autistic disorder from other developmental disorders and normal behaviour. Trained examiners scored videotapes of ADOS interactions, and reliability computed on 16% of the boys was 0.89 for the individual items (range 0.83–0.96) and 0.93 on diagnosis (range 0.81–1.00).</p> <hd id="AN0024165157-15">Maternal education</hd> <p>Maternal education was measured by total years of education at time of enrollment in the study and was included as a proxy for the effects of environmental factors. Evidence suggests that children with higher levels of maternal education have more sophisticated speech and language development ([<reflink idref="bib18" id="ref52">18</reflink>]; [<reflink idref="bib42" id="ref53">42</reflink>]; [<reflink idref="bib9" id="ref54">9</reflink>]; [<reflink idref="bib25" id="ref55">25</reflink>]). Maternal education levels at the first assessment are reported in Table 1.</p> <p>1 Means and standard deviations for TACL‐3 age equivalent scores and background variables</p> <p> <ephtml> <table><thead valign="bottom"><tr><th /><th><bold>Typically developing</bold></th><th><bold>FXS‐Only</bold></th><th><bold>FXS‐Spec</bold></th><th><bold>FXS‐Aut</bold></th><th><bold>Down syndrome</bold></th></tr><tr><th><bold><italic>n</italic></bold></th><th><bold>Mean (SD)</bold></th><th><bold><italic>n</italic></bold></th><th><bold>Mean (SD)</bold></th><th><bold><italic>N</italic></bold></th><th><bold>Mean (SD)</bold></th><th><bold><italic>n</italic></bold></th><th><bold>Mean (SD)</bold></th><th><bold><italic>N</italic></bold></th><th><bold>Mean (SD)</bold></th></tr></thead><tbody valign="top"><tr><td>Child age*</td></tr><tr><td> Visit 1</td><td>40</td><td> 66.2 (15.7)</td><td>35</td><td>128.4 (34.1)</td><td>24</td><td>112.7 (32.9)</td><td>19</td><td>120.5 (35.0)</td><td>45</td><td>114.6 (34.2)</td></tr><tr><td> Visit 2</td><td>32</td><td> 77.4 (14.7)</td><td>29</td><td>145.8 (30.5)</td><td>17</td><td>135.2 (33.2)</td><td>11</td><td>141.3 (36.2)</td><td>19</td><td>126.5 (31.9)</td></tr><tr><td> Visit 3</td><td> 1</td><td>115</td><td>11</td><td>150.3 (18.7)</td><td> 5</td><td>162.5 (18.3)</td><td> 3</td><td>152.2 (37.5)</td><td> 1</td><td>159.2</td></tr><tr><td>Vocab AE†</td></tr><tr><td> Visit 1</td><td>40</td><td> 73.7 (21.8)</td><td>34</td><td>56.3 (15.3)</td><td>22</td><td> 52.5 (12.3)</td><td>17</td><td> 47.1 (13.2)</td><td>37</td><td> 50.3 (13)</td></tr><tr><td> Visit 2</td><td>32</td><td> 92.5 (19.6)</td><td>29</td><td>59.7 (15.8)</td><td>17</td><td> 59.3 (15.2)</td><td>11</td><td> 54.3 (13.7)</td><td>19</td><td> 57.2 (15.2)</td></tr><tr><td> Visit 3</td><td> 1</td><td>117</td><td>11</td><td>64.9 (21.7)</td><td> 5</td><td> 56.4 (16.9)</td><td> 3</td><td> 61.0 (13.5)</td><td> 1</td><td> 54</td></tr><tr><td>ElabPhr AE‡</td></tr><tr><td> Visit 1</td><td>39</td><td> 78.2 (22.2)</td><td>32</td><td>55.2 (10.7)</td><td>22</td><td> 51.6 (10.5)</td><td>13</td><td> 48.0 (9.9)</td><td>43</td><td> 49.5 (13.7)</td></tr><tr><td> Visit 2</td><td>32</td><td> 90.6 (20.9)</td><td>28</td><td>55.7 (12.8)</td><td>14</td><td> 56.6 (12.7)</td><td> 8</td><td> 55.1 (14.0)</td><td>18</td><td> 60.2 (14.3)</td></tr><tr><td> Visit 3</td><td> 1</td><td>117</td><td>11</td><td>60.8 (18.8)</td><td> 5</td><td> 55.8 (14.5)</td><td> 2</td><td> 73.5 (23.3)</td><td> 1</td><td> 66</td></tr><tr><td>GraMo AE§</td></tr><tr><td> Visit 1</td><td>39</td><td> 77.7 (22.2)</td><td>31</td><td>60.5 (12.4)</td><td>21</td><td> 54.7 (13.3)</td><td>16</td><td> 53.6 (14.2)</td><td>35</td><td> 50.3 (12)</td></tr><tr><td> Visit 2</td><td>32</td><td> 85.9 (20.5)</td><td>27</td><td>63.4 (13.0)</td><td>14</td><td> 55.9 (11.4)</td><td> 9</td><td> 63.0 (19.2)</td><td>19</td><td> 55.1 (13.1)</td></tr><tr><td> Visit 3</td><td> 1</td><td>117</td><td>11</td><td>70.1 (16.8)</td><td> 5</td><td> 65.4 (16.8)</td><td> 3</td><td> 55.0 (14.8)</td><td> 1</td><td> 66</td></tr><tr><td>Leiter‐R AE¶</td></tr><tr><td> Visit 1</td><td>40</td><td> 70.2 (20)</td><td>35</td><td>63.0 (7.8)</td><td>24</td><td> 62.5 (9.0)</td><td>19</td><td> 54.4 (10.3)</td><td>45</td><td> 57.7 (12.2)</td></tr><tr><td> Visit 2</td><td>32</td><td> 85.5 (18.3)</td><td>29</td><td>65.5 (7.3)</td><td>17</td><td> 62.4 (5.7)</td><td>11</td><td> 60.9 (5.2)</td><td>19</td><td> 67.7 (10.9)</td></tr><tr><td> Visit 3</td><td> 1</td><td>120</td><td>11</td><td>68.1 (9.4)</td><td> 5</td><td> 67.2 (5.8)</td><td> 3</td><td> 62.0 (3.6)</td><td> 1</td><td> 64</td></tr><tr><td>Maternal education**</td><td>40</td><td> 16.4 (2.2)</td><td>35</td><td>14.2 (2.3)</td><td>24</td><td> 14.5 (1.9)</td><td>19</td><td> 15.4 (2.5)</td><td>45</td><td> 15.5 (2.4)</td></tr></tbody></table> </ephtml> </p> <p>1 * Chronological age is age in months at TACL‐3 testing.</p> <ulist> <item>2 † Vocabulary subtest age equivalent.</item> <item>3 ‡ Elaborated Phrases and Sentences subtest age equivalent.</item> <item>4 § Grammatical Morphemes subtest age equivalent.</item> <item>5 ¶ Leiter‐R age equivalent.</item> <item>6 ** Maternal education is years of school completed at time of first TACL‐3 assessment.</item> <item>7 TACL‐3, Test for Auditory Comprehension of Language – 3rd Edition; FXS, fragile X syndrome; FXS‐Spec, fragile X syndrome and autism spectrum; FXS‐Aut, fragile X syndrome and autism; Leiter‐R, Leiter International Performance Scale – Revised.</item> </ulist> <hd id="AN0024165157-16">Analysis strategy</hd> <p>The analysis model reported here is a hierarchical linear model (HLM) with multiple observations for each child in the study. HLM was used so that we could model both the within‐child variability and between‐child variability. The question of interest was whether the TD boys, boys with FXS‐Only, boys with FXS‐Aut, boys with FXS‐Spec and boys with DS showed similar patterns of receptive vocabulary, grammatical morphology and syntax skills while maternal education and non‐verbal cognition were controlled for [Leiter‐R developmental age (DA)]. The outcome measures were the three subtests of the TACL‐3: Vocabulary, Grammatical Morphemes, and Elaborated Phrases and Sentences. Maternal education level and Leiter‐R DA were mean centred.</p> <p>The analysis model contained main effects for TACL−3 subtests and syndrome group with maternal education and Leiter‐R DA as covariates. We tested for two‐way interactions between subtest and syndrome group, subtest and maternal education, subtest and Leiter‐R DA, syndrome group and maternal education, and syndrome group and Leiter‐R DA. The two‐way interactions were not significant. We also tested for three‐way interactions between subtest, syndrome group and maternal education, and between subtest, syndrome group and Leiter‐R DA. These three‐way interactions were non‐significant both individually and as a block and were removed from the final model.</p> <hd id="AN0024165157-17">Results</hd> <p></p> <hd id="AN0024165157-18">Variability among children</hd> <p>Table 1 provides descriptive information about receptive language skills for each assessment across the five groups. All groups demonstrated considerable individual variability. As shown in Table 2, TACL‐3 scores were moderately to highly correlated with each other and with Leiter‐R scores for the TD and DS groups. The relationships between Leiter‐R and TACL‐3 scores were generally lower for the FXS groups.</p> <p>2 Correlations among TACL‐3 subtests and Leiter‐R developmental age at assessment 1</p> <p> <ephtml> <table><thead valign="bottom"><tr><th /><th><bold>GraMo</bold></th><th><bold>ElabPhr</bold></th><th><bold>Leiter‐R</bold></th></tr></thead><tbody valign="top"><tr><td>TD</td></tr><tr><td> Vocab†</td><td>0.74***</td><td>0.71***</td><td>0.75***</td></tr><tr><td> GraMo‡</td><td /><td>0.79***</td><td>0.68***</td></tr><tr><td> ElabPhr§</td><td /><td /><td>0.68***</td></tr><tr><td>DS</td></tr><tr><td> Vocab†</td><td>0.60***</td><td>0.81***</td><td>0.60***</td></tr><tr><td> GraMo‡</td><td /><td>0.67***</td><td>0.70***</td></tr><tr><td> ElabPhr§</td><td /><td /><td>0.64***</td></tr><tr><td>FXS‐only</td></tr><tr><td> Vocab†</td><td>0.59***</td><td>0.51**</td><td>0.49**</td></tr><tr><td> GraMo‡</td><td /><td>0.56**</td><td>0.35</td></tr><tr><td> ElabPhr§</td><td /><td /><td>0.39*</td></tr><tr><td>FXS‐Spec</td></tr><tr><td> Vocab†</td><td>0.61**</td><td>0.36</td><td>0.56**</td></tr><tr><td> GraMo‡</td><td /><td>0.45*</td><td>0.58**</td></tr><tr><td> ElabPhr§</td><td /><td /><td>0.35</td></tr><tr><td>FXS‐Aut</td></tr><tr><td> Vocab†</td><td>0.81***</td><td>0.59*</td><td>0.64**</td></tr><tr><td> GraMo‡</td><td /><td>0.69*</td><td>0.56*</td></tr><tr><td> ElabPhr§</td><td /><td /><td>0.65*</td></tr></tbody></table> </ephtml> </p> <ulist> <item>8 * <emph> P</emph> < 0.05;</item> <item>9 ** <emph> P</emph> < 0.01;</item> <item>10 ***  <emph>P</emph> < 0.001.</item> <item>11 † Vocabulary subtest age equivalent.</item> <item>12 ‡  Grammatical Morphemes subtest age equivalent.</item> <item>13 § Elaborated Phrases and Sentences subtest age equivalent.</item> <item>14 TD, typically developing; DS, Down syndrome; TACL‐3, Test for Auditory Comprehension of Language – 3rd Edition; FXS, fragile X syndrome; FXS‐Spec, fragile X syndrome and autism spectrum; FXS‐Aut, fragile X syndrome and autism; Leiter‐R, Leiter International Performance Scale – Revised.</item> </ulist> <hd id="AN0024165157-19">Test for Auditory Comprehension of Language – 3rd Edition analysis</hd> <p>Table 3 lists the statistical tests, parameter estimates, their standard errors and the adjusted means where they are applicable. There were significant main effects for syndrome group (<emph>F</emph>(<reflink idref="bib4" id="ref56">4</reflink>,<reflink idref="bib153" id="ref57">153</reflink>) = 28.66, <emph>P</emph> < 0.0001), Leiter‐R DA (<emph>F</emph>(<reflink idref="bib1" id="ref58">1</reflink>,<reflink idref="bib630" id="ref59">630</reflink>) = 137.54, <emph>P</emph> < 0.0001) and maternal education (<emph>F</emph>(<reflink idref="bib1" id="ref60">1</reflink>,<reflink idref="bib153" id="ref61">153</reflink>) = 5.79, <emph>P</emph> = 0.02). There were no significant effects for subtests or for any of the interaction terms. The lack of significant interactions indicates that there was no differential performance because of any of the covariates between the subtests.</p> <p>3 Mixed model results and adjusted means for TACL‐3 analysis</p> <p> <ephtml> <table><thead valign="bottom"><tr><th><bold>Effect</bold></th><th><bold>Test</bold></th><th><bold>Subtest</bold></th><th><bold>Group</bold></th><th><bold>B (SE)</bold></th><th><bold>M</bold></th></tr></thead><tbody valign="top"><tr><td>Intercept</td><td /><td /><td /><td> 70.87*** (2.02)</td><td /></tr><tr><td>Subtest</td><td><italic>F</italic>(2,289) = 0.86</td><td>Vocab†</td><td /><td>  0</td><td /></tr><tr><td>GraMo‡</td><td /><td>  1.86 (2.81)</td><td /></tr><tr><td>ElabPhr§</td><td /><td>  3.29 (2.75)</td><td /></tr><tr><td>Syndrome group</td><td><italic>F</italic>(4,153) = 28.66***</td><td /><td>TD</td><td /><td>73.49†</td></tr><tr><td>FXS‐Only</td><td> −9.82*** (2.85)</td><td>61.18‡</td></tr><tr><td>FXS‐Spec</td><td>−11.49*** (3.19)</td><td>59.28‡<sup>,</sup>§</td></tr><tr><td>FXS‐Aut</td><td>−13.79*** (3.73)</td><td>58.56‡<sup>,</sup>§</td></tr><tr><td>DS</td><td>−16.08*** (2.83)</td><td>55.46§</td></tr><tr><td>Leiter‐R developmental age</td><td><italic>F</italic>(1,630) = 137.54***</td><td /><td /><td>  0.71*** (0.06)</td><td /></tr><tr><td>Maternal education</td><td><italic>F</italic>(1,153) = 5.79*</td><td /><td /><td>  0.64* (0.27)</td><td /></tr><tr><td>Syndrome group × maternal education</td><td><italic>F</italic>(4,153) = 2.19</td><td /><td /><td /><td /></tr><tr><td>Syndrome group × Leiter‐R developmental age</td><td><italic>F</italic>(4,630) = 0.69</td><td /><td /><td /><td /></tr><tr><td>Subtest × Leiter‐R developmental age</td><td><italic>F</italic>(2,630) = 2.70</td><td /><td /><td /><td /></tr><tr><td>Subtest × maternal education</td><td><italic>F</italic>(2,630) = 0.61</td><td /><td /><td /><td /></tr><tr><td>Subtest × syndrome group</td><td><italic>F</italic>(8,289) = 0.92</td><td /><td /><td /><td /></tr></tbody></table> </ephtml> </p> <ulist> <item>15 * <emph> P</emph> < 0.05;</item> <item>16 **  <emph>P</emph> < 0.01;</item> <item>17 *** <emph> P</emph> < 0.001.</item> <item>18 B are regression coefficients, SE are standard errors and M are adjusted means. Means with different superscripts are significantly different.</item> <item>19 † Vocabulary subtest age equivalent.</item> <item>20 ‡  Grammatical morphemes subtest age equivalent.</item> <item>21 § Elaborated phrases subtest age equivalent.</item> <item>22 TD, typically developing; DS, Down syndrome; TACL‐3, Test for Auditory Comprehension of Language – 3rd Edition; FXS, fragile X syndrome; FXS‐Spec, fragile X syndrome and autism spectrum; FXS‐Aut, fragile X syndrome and autism; Leiter‐R, Leiter International Performance Scale – Revised.</item> </ulist> <p>Although there was no differential performance by group over the different TACL‐3 subtests, there were differences between the syndrome groups in how they did on the TACL‐3 on average. Boys with DS scored lower (M = 55.5) than the boys with FXS‐Only (M = 61.2) but not significantly different from boys with FXS‐Spec (M = 59.3) or FXS‐Aut (M = 58.6). Boys with FXS did not differ significantly from each other according to autism status. TD boys (M = 73.5) scored higher than all of the other groups. In addition, we found that as non‐verbal cognition (Leiter‐R DA) increased, the TACL‐3 score increased by about 0.71 month for every month above the mean Leiter‐R DA. Increases in maternal education had a similar positive effect. For every year of maternal education above the mean, the TACL‐3 score will increase by about 0.64 points. Neither of these covariates has differential effects over the TACL‐3 subtests.</p> <hd id="AN0024165157-20">Discussion</hd> <p>After controlling for non‐verbal cognition and maternal education, we found that the language comprehension skills of boys with FXS, regardless of autism status, and boys with DS were lower than those of TD boys. Boys with FXS did not differ from each other based on their autism status. Boys with FXS‐Only scored higher than boys with DS. For all of the groups, scores across the Vocabulary, Grammatical Morphemes, and Elaborated Phrases and Sentences subtests of the TACL‐3 did not differ<emph>.</emph></p> <p>Our finding that boys with FXS scored lower than TD matches after controlling for non‐verbal cognition and maternal education differs from previous reports of individuals with FXS in which language comprehension levels were similar to those of non‐verbal cognitive matches ([<reflink idref="bib39" id="ref62">39</reflink>]; [<reflink idref="bib3" id="ref63">3</reflink>]). Our participants, with a mean age of about 10 years, were younger than those in [<reflink idref="bib3" id="ref64">3</reflink>]) and [<reflink idref="bib39" id="ref65">39</reflink>]) studies. Research on other populations with ID has indicated that receptive vocabulary levels are related to chronological age ([<reflink idref="bib24" id="ref66">24</reflink>]; [<reflink idref="bib38" id="ref67">38</reflink>]). Moreover, as suggested by [<reflink idref="bib38" id="ref68">38</reflink>]), individuals with ID may have greater difficulty on the TACL‐3, which measures comprehension of conceptually difficult items, as compared to measures such as the Peabody Picture Vocabulary Test ([<reflink idref="bib20" id="ref69">20</reflink>]; [<reflink idref="bib21" id="ref70">21</reflink>]) used by [<reflink idref="bib35" id="ref71">35</reflink>]) which measures experience‐related vocabulary knowledge. We also note that [<reflink idref="bib3" id="ref72">3</reflink>]) sample included girls, who are less severely affected by FXS than boys ([<reflink idref="bib28" id="ref73">28</reflink>]), while our participants were all boys.</p> <p>Our results indicate that the receptive language of boys with FXS does not differ according to autism status after controlling for non‐verbal cognition and maternal education, in contrast to the findings of [<reflink idref="bib46" id="ref74">46</reflink>]) and [<reflink idref="bib40" id="ref75">40</reflink>]). However, [<reflink idref="bib46" id="ref76">46</reflink>]) and [<reflink idref="bib40" id="ref77">40</reflink>]) studies examined younger children and used a single broad measure of receptive language. Furthermore, in Rogers <emph>et al</emph>.'s study, the groups did not differ significantly on overall mental age or socio‐economic status; however, non‐verbal cognition was not controlled for in the analyses. In the Philofsky study, groups differed significantly on non‐verbal mental age, and this variable was not controlled for in the analyses. Our findings agreed with those of Roberts <emph>et al</emph>. (manuscript under review), who found that receptive vocabulary levels of boys with FXS did not differ according to autism status when non‐verbal cognition and maternal education levels were controlled.</p> <p>Our participants with DS scored lower than non‐verbal mental age matches in receptive vocabulary. This finding differs from previous literature in which the Peabody Picture Vocabulary Test and British Picture Vocabulary Scale – Second Edition ([<reflink idref="bib22" id="ref78">22</reflink>]) were used to measure receptive vocabulary ([<reflink idref="bib12" id="ref79">12</reflink>]; [<reflink idref="bib30" id="ref80">30</reflink>]). Again, the relatively young age of our participants and our use of the TACL‐3 may have influenced our findings regarding receptive vocabulary. Our finding that the receptive morphology and syntax skills of boys with DS were lower than those of TD non‐verbal cognitive matches is consistent with previous literature ([<reflink idref="bib48" id="ref81">48</reflink>]; [<reflink idref="bib14" id="ref82">14</reflink>]; [<reflink idref="bib30" id="ref83">30</reflink>]). Our findings also indicate that the language comprehension skills of boys with DS are lower than those of boys with FXS‐Only, agreeing with the findings of [<reflink idref="bib3" id="ref84">3</reflink>]) and Roberts <emph>et al</emph>. (manuscript under review).</p> <p>We also found that receptive language skills were generally correlated with non‐verbal cognitive levels for all groups, similar to the findings of previous studies of individuals with FXS and DS ([<reflink idref="bib12" id="ref85">12</reflink>]; [<reflink idref="bib3" id="ref86">3</reflink>]). This supports social interactionist perspectives of language ([<reflink idref="bib7" id="ref87">7</reflink>]; [<reflink idref="bib34" id="ref88">34</reflink>]; [<reflink idref="bib15" id="ref89">15</reflink>]; [<reflink idref="bib1" id="ref90">1</reflink>]), which emphasize the bidirectional influence of linguistic and non‐linguistic development. Interestingly, language comprehension and non‐verbal cognition were less highly correlated for boys with FXS than for the other groups, suggesting that linguistic and non‐linguistic development, while related, may not be as closely linked in individuals with FXS.</p> <p>The strengths of this study include our relatively large sample sizes and the fact that we controlled for boys' non‐verbal cognition and maternal education, factors that have been shown previously to affect language development. In addition, we prospectively documented language comprehension skills with up to three assessments. Other child factors such as working memory and FMRP levels (for boys with FXS) and maternal factors such as responsivity and genetic status (for mothers of boys with FXS) may also be important predictors of language comprehension skills and should be examined in future studies.</p> <hd id="AN0024165157-21">Clinical implications</hd> <p>Language comprehension skills appear to be an important area to target in assessment and intervention for both boys with FXS and boys with DS. Because performance across measures of vocabulary, grammatical morphology, and elaborated phrases and sentences did not vary for any of the groups, intervention in all three of these areas may be equally important. Given the wide variability in the development of language comprehension for all of the groups, each child should receive a thorough language assessment, and the intervention plan should reflect each child's unique strengths and weaknesses. 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Items – Name: Title
  Label: Title
  Group: Ti
  Data: Language Comprehension in Boys with Fragile X Syndrome and Boys with Down Syndrome
– Name: Language
  Label: Language
  Group: Lang
  Data: English
– Name: Author
  Label: Authors
  Group: Au
  Data: <searchLink fieldCode="AR" term="%22Price%2C+J%2E%22">Price, J.</searchLink><br /><searchLink fieldCode="AR" term="%22Roberts%2C+J%2E%22">Roberts, J.</searchLink><br /><searchLink fieldCode="AR" term="%22Vandergrift%2C+N%2E%22">Vandergrift, N.</searchLink><br /><searchLink fieldCode="AR" term="%22Martin%2C+G%2E%22">Martin, G.</searchLink>
– Name: TitleSource
  Label: Source
  Group: Src
  Data: <searchLink fieldCode="SO" term="%22Journal+of+Intellectual+Disability+Research%22"><i>Journal of Intellectual Disability Research</i></searchLink>. Apr 2007 51(4):318-326.
– Name: Avail
  Label: Availability
  Group: Avail
  Data: Blackwell Publishing. 350 Main Street, Malden, MA 02148. Tel: 800-835-6770; Tel: 781-388-8599; Fax: 781-388-8232; e-mail: customerservices@blackwellpublishing.com; Web site: http://www.blackwellpublishing.com/jnl_default.asp
– Name: PeerReviewed
  Label: Peer Reviewed
  Group: SrcInfo
  Data: Y
– Name: Pages
  Label: Page Count
  Group: Src
  Data: 9
– Name: DatePubCY
  Label: Publication Date
  Group: Date
  Data: 2007
– Name: TypeDocument
  Label: Document Type
  Group: TypDoc
  Data: Journal Articles<br />Reports - Research
– Name: Subject
  Label: Descriptors
  Group: Su
  Data: <searchLink fieldCode="DE" term="%22Morphology+%28Languages%29%22">Morphology (Languages)</searchLink><br /><searchLink fieldCode="DE" term="%22Down+Syndrome%22">Down Syndrome</searchLink><br /><searchLink fieldCode="DE" term="%22Syntax%22">Syntax</searchLink><br /><searchLink fieldCode="DE" term="%22Sentences%22">Sentences</searchLink><br /><searchLink fieldCode="DE" term="%22Receptive+Language%22">Receptive Language</searchLink><br /><searchLink fieldCode="DE" term="%22Males%22">Males</searchLink><br /><searchLink fieldCode="DE" term="%22Listening+Comprehension%22">Listening Comprehension</searchLink><br /><searchLink fieldCode="DE" term="%22Language+Skills%22">Language Skills</searchLink><br /><searchLink fieldCode="DE" term="%22Autism%22">Autism</searchLink><br /><searchLink fieldCode="DE" term="%22Genetics%22">Genetics</searchLink><br /><searchLink fieldCode="DE" term="%22Congenital+Impairments%22">Congenital Impairments</searchLink><br /><searchLink fieldCode="DE" term="%22Mental+Retardation%22">Mental Retardation</searchLink><br /><searchLink fieldCode="DE" term="%22Grammar%22">Grammar</searchLink><br /><searchLink fieldCode="DE" term="%22Pervasive+Developmental+Disorders%22">Pervasive Developmental Disorders</searchLink><br /><searchLink fieldCode="DE" term="%22Comparative+Analysis%22">Comparative Analysis</searchLink><br /><searchLink fieldCode="DE" term="%22Vocabulary%22">Vocabulary</searchLink>
– Name: SubjectThesaurus
  Label: Assessment and Survey Identifiers
  Group: Su
  Data: <searchLink fieldCode="SU" term="%22Test+for+Auditory+Comprehension+of+Language%22">Test for Auditory Comprehension of Language</searchLink>
– Name: DOI
  Label: DOI
  Group: ID
  Data: 10.1111/j.1365-2788.2006.00881.x
– Name: ISSN
  Label: ISSN
  Group: ISSN
  Data: 0964-2633
– Name: Abstract
  Label: Abstract
  Group: Ab
  Data: Background: Fragile X syndrome (FXS) is the most common known inherited cause of intellectual disability, yet very few studies have explored the language comprehension skills of children with FXS. We examined the receptive vocabulary, grammatical morphology and syntax skills of boys with FXS (who were additionally classified as having autism, autism spectrum, or no autism) and compared them to boys with Down syndrome (DS) and typically developing (TD) boys at similar non-verbal developmental levels. Methods: The Vocabulary, Grammatical Morphology, and Elaborated Phrases and Sentences subtests of the Test for Auditory Comprehension of Language--3rd Edition (TACL-3) were administered annually up to three times to assess the language comprehension skills of 35 boys with FXS without autism, 24 boys with FXS with autism spectrum, 19 boys with FXS with autism, 45 boys with DS and 40 TD boys at similar non-verbal cognitive levels. Results: After controlling for non-verbal cognition and maternal education levels, we found that the three groups of boys with FXS did not differ from each other but scored lower than the TD boys in language comprehension. The boys with DS scored lower in language comprehension than boys with FXS without autism and TD boys. For all of the groups, scores for receptive vocabulary, grammatical morphology and syntax did not differ. Conclusions: Boys with FXS and boys with DS differed in receptive language levels, demonstrating unique language profiles for each syndrome. Language comprehension appears to be an important area to target in assessment and intervention for both populations.
– Name: AbstractInfo
  Label: Abstractor
  Group: Ab
  Data: Author
– Name: Ref
  Label: Number of References
  Group: RefInfo
  Data: 51
– Name: DateEntry
  Label: Entry Date
  Group: Date
  Data: 2007
– Name: AN
  Label: Accession Number
  Group: ID
  Data: EJ754595
PLink https://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=eric&AN=EJ754595
RecordInfo BibRecord:
  BibEntity:
    Identifiers:
      – Type: doi
        Value: 10.1111/j.1365-2788.2006.00881.x
    Languages:
      – Text: English
    PhysicalDescription:
      Pagination:
        PageCount: 9
        StartPage: 318
    Subjects:
      – SubjectFull: Morphology (Languages)
        Type: general
      – SubjectFull: Down Syndrome
        Type: general
      – SubjectFull: Syntax
        Type: general
      – SubjectFull: Sentences
        Type: general
      – SubjectFull: Receptive Language
        Type: general
      – SubjectFull: Males
        Type: general
      – SubjectFull: Listening Comprehension
        Type: general
      – SubjectFull: Language Skills
        Type: general
      – SubjectFull: Autism
        Type: general
      – SubjectFull: Genetics
        Type: general
      – SubjectFull: Congenital Impairments
        Type: general
      – SubjectFull: Mental Retardation
        Type: general
      – SubjectFull: Grammar
        Type: general
      – SubjectFull: Pervasive Developmental Disorders
        Type: general
      – SubjectFull: Comparative Analysis
        Type: general
      – SubjectFull: Vocabulary
        Type: general
      – SubjectFull: Test for Auditory Comprehension of Language
        Type: general
    Titles:
      – TitleFull: Language Comprehension in Boys with Fragile X Syndrome and Boys with Down Syndrome
        Type: main
  BibRelationships:
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            NameFull: Price, J.
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            NameFull: Roberts, J.
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            NameFull: Vandergrift, N.
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            NameFull: Martin, G.
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              Type: published
              Y: 2007
          Identifiers:
            – Type: issn-print
              Value: 0964-2633
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            – Type: volume
              Value: 51
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            – TitleFull: Journal of Intellectual Disability Research
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