PEDIATRICS Vol. 118 No. 5 November 2006, pp. e1541-e1549 (doi:10.1542/peds.2005-2761)
ARTICLE |
The Clinical Spectrum of Developmental Language Impairment in School-Aged Children: Language, Cognitive, and Motor Findings
a Department of Neurology/Neurosurgery
c School of Communications Sciences and Disorders
d Department of Pediatrics
e School of Physical and Occupational Therapy
f McConnell Brain Imaging Centre, McGill University, Montreal, Quebec, Canada
b Department of Neurology and Children's Hospital Education Research Institute, Children's Hospital at Westmead, Westmead, New South Wales, Australia
| ABSTRACT |
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OBJECTIVE. Our goal was to evaluate detailed school-age language, nonverbal cognitive, and motor development in children with developmental language impairment compared with age-matched controls.
METHODS. Children with developmental language impairment or normal language development (controls) aged 7 to 13 years were recruited. Children underwent language assessment (Clinical Evaluation of Language Fundamentals-4, Peabody Picture Vocabulary-3, Goldman-Fristoe Test of Articulation-2), nonverbal cognitive assessment (Wechsler Intelligence Scale for Children-IV), and motor assessment (Movement Assessment Battery for Children). Exclusion criteria were nonverbal IQ below the 5th percentile or an acquired language, hearing, autistic spectrum, or neurologic disorder.
RESULTS. Eleven children with developmental language impairment (7:4 boys/girls; mean age: 10.1 ± 0.8 years) and 12 controls (5:7 boys/girls; mean age: 9.5 ± 1.8 years) were recruited. Children with developmental language impairment showed lower mean scores on language (Clinical Evaluation of Language Fundamentals-4—developmental language impairment: 79.7 ± 16.5; controls: 109.2 ± 9.6; Goldman-Fristoe Test of Articulation-2—developmental language impairment: 94.1 ± 10.6; controls: 104.0 ± 2.8; Peabody Picture Vocabulary-3—developmental language impairment: 90.5 ± 13.8; controls: 100.1 ± 11.6), cognitive (Wechsler Intelligence Scale for Children-IV—developmental language impairment: 99.5 ± 15.5; controls: 113.5 ± 11.9), and motor measures (Movement Assessment Battery for Children percentile—developmental language impairment: 12.7 ± 16.7; controls: 66.1 ± 30.6) and greater discrepancies between cognitive and language scores (Wechsler Intelligence Scale for Children-IV/Clinical Evaluation of Language Fundamentals-4—developmental language impairment: 17.8 ± 17.8; controls: 1.2 ± 12.7). Motor impairment was more common in children with developmental language impairment (70%) than controls (8%).
CONCLUSIONS. Developmental language impairment is characterized by a broad spectrum of developmental impairments. Children identified on the basis of language impairment show significant motor comorbidity. Motor assessment should form part of the evaluation and follow-up of children with developmental language impairment.
Key Words: cognitive development language disorders developmental delay motor development language development
Abbreviations: SLI—specific language impairment DLI—developmental language impairment CELF-4—Clinical Evaluation of Language Fundamentals-4 PPVT-3—Peabody Picture Vocabulary Test, 3rd Edition GFTA-2—Goldman-Fristoe Test of Articulation-2 WISC-IV—Wechsler Intelligence Scale for Children, 4th Edition PRI—Perceptual Reasoning Index M-ABC—Movement Assessment Battery for Children
Developmental disorders leading to language impairment are probably the most common form of childhood developmental disability. The prevalence of language delay in preschool-aged children has been estimated to be 7.6%.1 In kindergarten-aged children, 7.4% were found to meet criteria for specific language impairment (SLI).2,3 The term SLI has been used to identify children with language impairment in the context of normal nonverbal cognitive function; however, evidence is increasing that SLI is associated with a range of impairments in other developmental domains.4–6 Thus, in this article, we use the term developmental language impairment (DLI) to describe children who would otherwise meet criteria for SLI. Children with an acquired language disorder or language impairment secondary to an autistic spectrum disorder, a hearing disorder, or a known neurologic disorder are usually not considered to have DLI.
Previous studies that examined the clinical phenotypes of children with DLI have identified a range of impairments in domains other than language. There is an increased incidence of attention-deficit disorders among children with DLI7 and an increased incidence of language impairment among children with attention-deficit disorders.5 Children with DLI are frequently found to have impairments in socialization skills.8 Moreover, despite the requirement for normal nonverbal cognitive function, nonverbal cognitive impairments have been reported at school-age follow-up in children with a diagnosis of DLI made at preschool age.9 Despite the range of impairments seen in children with DLI, it is unclear whether these deficits are secondary to the effects over time of the underlying communication disorder or whether they are a separate but intrinsic part of the underlying disorder that leads to language impairment.
There is increasing evidence that motor impairment is a common comorbidity in children with DLI.4,6,10,11 Motor impairment is less likely to be secondary to a communication disorder than the impairments in other developmental domains. Among children with DLI, motor impairment has been found to correlate most strongly with the observed severity of the child's language disorder.5 Consistent with this, in studies that have recruited children with greater degrees of language impairment, a higher incidence of motor disorders has been reported.12 However, it is still unclear whether motor impairment in children with DLI is the result of a more global developmental impairment or reflects a biological function that has greater effects on language and motor function than nonverbal cognition.
Most previous studies that have evaluated motor performance in DLI have used measures of isolated motor functions (eg, rate of tapping or peg-moving10,13,14) or have used combined measures of neurologic soft signs that are not standardized and do not necessarily predict a child's functional motor ability.15,16 Four studies have used standardized measures of motor function to evaluate children with DLI.4,6,11,12 In one of these studies a proportion of children would not meet criteria for DLI,12 and the other studies focused on the motor findings of children with DLI but provided limited data on the children's language and cognitive skills.4,6,11 To our knowledge, no study has evaluated the developmental profiles of children with DLI across language, nonverbal cognitive, and motor domains.
With this study we aimed to further the understanding of DLI by evaluating the developmental profiles (ie, phenotypes) of children with DLI across language, motor, and nonverbal cognitive domains using standardized instruments with relevance for clinicians.
| METHODS |
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Population
Children between the ages of 7 and 13 years who were considered to have DLI and controls were prospectively recruited for this study. Children were recruited through the neurology and developmental clinics of Montreal Children's Hospital, private speech/language pathologists, and from a class for children with language disorders. For the DLI group, clinicians were asked to refer children if they had an impairment in language but were considered to have otherwise-normal nonverbal cognitive development. For the control group, clinicians were asked to refer children with headache disorders (ie, migraines, considered unlikely to have a structural basis) who were considered to have normal development in all developmental domains. Children with headache disorders were selected as the comparison group because these children are regularly assessed through the hospital's neurology clinics. These children had already undergone assessment by a neurologist and, thus, were unlikely to have unrecognized developmental problems.
Given that the normative data for the language and cognitive measures were available for English-speaking children, only children with English as their dominant language were recruited.
Children were excluded from this study if they were known to have an underlying neurologic or autistic spectrum disorder per Diagnostic and Statistical Manual of Mental Disorders, Fourth Edition, Revised criteria (ie, pervasive developmental disorder or pervasive developmental disorder-not otherwise specified), a hearing impairment, or on subsequent testing were found to have a nonverbal IQ below the 5th percentile. Approval was obtained from the hospital's institutional review board before commencing the study. Written informed consent from the children's parents and the child's assent were necessary preconditions for participation in this study.
Clinical Assessment and Measures Used
Before assessment, the parents of all children were interviewed by telephone by a research assistant to confirm that they met broad eligibility criteria. All children underwent screening audiometry with a portable audiometer. A threshold of 20 dB (at least in 1 ear) at all frequencies tested was considered to represent adequate hearing for conversational speech. A neurologic examination was performed by a pediatric neurologist to identify signs suggestive of possible underlying structural neurologic disease. Parents were asked to complete a questionnaire providing details about the child's medical and developmental history as well as the Pragmatics Profile from the Clinical Evaluation of Language Fundamentals-4 (CELF-4).17 All speech/language, psychological, and occupational therapy assessments were performed by appropriately qualified therapists who were blinded to the children's group assignment (suspected DLI or control) and to clinical information.
Language was assessed by using the CELF-4 and the Peabody Picture Vocabulary Test, 3rd Edition (PPVT-3).18 The CELF-4 is an instrument that was designed to identify language disorders or delays in children >5 years of age. The CELF-4 shows excellent split-half and interrater reliability, and detailed evidence supporting its validity is presented in its published manual.17 The PPVT-3 is a well-established measure of receptive vocabulary for children and adults aged >2 years. It shows excellent internal consistency and test-retest reliability, and validity is supported by strong correlations with other measures of language.18 The core subtests and the Pragmatics Profile of the CELF-4 were administered. The core subtests of the CELF-4 involve 2 tests of receptive language (concepts and following directions, word classes [receptive, for children >8 years old]) and 3 tests of expressive language (word structure [children <8 years old], word classes [children >8 years old], formulated sentences and recalling sentences). A parent-completed questionnaire, the Pragmatics Profile, was used to identify children who had evidence of pragmatic impairment (ie, the rules that determine how language is used in different social contexts and environments). Speech was assessed by using the "sounds-in-words" section of the Goldman-Fristoe Test of Articulation-2 (GFTA-2).19 This test is a systematic means of assessing each child's spontaneous articulation of the consonant sounds of standard American English in single words. The GFTA-2 has excellent test-retest, interrater, and internal reliability and is intended for use in children >2 years of age.19
The children's nonverbal cognition was assessed by using the block-design, matrix-reasoning, and picture-concepts subtests of the Wechsler Intelligence Scale for Children, 4th Edition (WISC-IV).20 These 3 subtests allow the calculation of a child's Perceptual Reasoning Index (PRI), which is a measure of nonverbal and fluid reasoning (the ability to deduce the relationship between stimuli and to draw conclusions from this information). It shows excellent reliability and convergent validity with the previously validated 3rd edition of the WISC.20
Children's motor function was measure by using the Movement Assessment Battery for Children (M-ABC).21 The M-ABC is a commonly used instrument for the identification of motor impairment in children and provides measures of movement competence and manual dexterity as well as ball skills and static and dynamic balance. The M-ABC represents a minor revision of a previously validated test of motor impairment (Test of Motor Impairment [Henderson Revision])22; this test had a minimum test-retest reliability for any item of 0.75 and a minimum interrater reliability of 0.70. A recent validation study of the M-ABC in Chinese preschool-aged children reported a mean intraclass correlation coefficient of 0.96 across items and a test-retest reliability of 0.77.23 The M-ABC has demonstrated validity in identifying motor impairments in at-risk24 populations and has previously been reported to be a useful instrument for detecting motor difficulties in children with SLI.4
Normative values were taken from the data set provided by the tests' publishers. The CELF-4, WISC-IV, and the GFTA-2 allow the calculation of standard scores (mean: 100; SD: 15). The M-ABC generates an age-standardized impairment score from which a child's percentile can be calculated.
Statistical Analysis
Statistical analysis was performed by using SPSS 11.5 software.25 Descriptive statistics were used to describe the population characteristics and the range of scores on the measures used. The distributions of scores are shown graphically by using box plots (see Figs 1–4). Comparisons between children with DLI and controls were performed by using t tests for normally distributed data (age at assessment, CELF-4 language scores, GFTA-2 scores, WISC-IV PRI scores, nonverbal cognitive discrepancy) and the Mann-Whitney tests (M-ABC impairment scores) for data that were not normally distributed. Given the difficulties inherent in comparing percentile ranks, statistical tests on the M-ABC were performed by using the total impairment score. To compare the distribution of scores with expected normative values, the sign test was used because of the relatively low sample size. The
2 statistic (or Fisher's exact test) was used to compare parental income brackets, the reported frequency of a family history of language disorders, and the frequency of motor impairments.
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| RESULTS |
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Thirty children (16 DLI, 14 controls) were originally recruited to participate in this study. Five children referred with a diagnosis of DLI were excluded from the study: 2 children did not undergo complete clinical testing, 1 child was found to have a PRI below the 5th percentile, 1 child had an acquired language disorder, and 1 child was considered to have normal language after assessment. Two other children (1 with DLI, 1 control) who met criteria for inclusion in the study but were the older siblings of children assessed were excluded to avoid biasing the data as a result of familial factors. Thus, 11 children with DLI (7 boys) and 12 controls (5 boys) met eligibility criteria for this study.
The demographic details and parental assessment of the children's attention, social, and reading skills for each child are presented in Table 1. A comparison of controls and children with DLI at the time of their clinical assessment is shown in Table 2. The age range of control children (9.5 ± 1.8 years) was more widely distributed than that of the children with DLI (10.1 ± 0.8 years), although the mean values did not differ significantly. Children with DLI were significantly more likely that control children to have a family history of language disorders (DLI: 6 of 11; controls: 0 of 10; P = .006), which is consistent with previous studies. All children passed screening audiometry. No child had clinical findings suggestive of a focal neurologic disorder (other than language impairment).
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All parents of children referred with a diagnosis of DLI considered their children to have problems with speech and language. The mother of 1 child in the control group reported that her child had difficulties with articulating some words. The majority of children (10 of 12) in the control group had a clinical diagnosis of migraine. Three children with DLI (3 of 11) had infrequent headaches; none were considered to have migraine. Data on the combined parental income were available for 9 children with DLI and 10 controls. The families of controls tended to have higher incomes than that of the families of children with DLI, although this did not reach statistical significance (P = .11).
The distributions of language (CELF-4), vocabulary (PPVT-3), and articulation (GFTA-2) scores for the DLI and control groups are shown in Figs 1–3. The test and subtest scores for the CELF-4 and the PPVT-3 for individual children are shown in Table 3. Children with DLI showed significantly lower total language scores on the CELF-4 than did controls (DLI: 79.7 ± 16.5; controls: 109.2 ± 9.6; P < .001). They also had significantly lower scores on tests of articulation (GFTA-2—DLI: 94.1 ± 10.6; controls: 104.0 ± 2.8; P = .003). Children with DLI tended to have poorer receptive vocabulary scores, although this did not reach statistical significance (PPVT-3—DLI: 90.5 ± 13.8; controls: 100.1 ± 11.6; P = .06). Children with DLI had significantly poorer scores on all the administered subtests of the CELF-4 other than word classes (P = .10). The subtest that caused the greatest difficulty was recalling sentences (DLI: 5.2 ± 3.2; controls: 10.9 ± 2.8; P = .001). Two children, both with a diagnosis of DLI, met the criterion for pragmatic impairment on the CELF-4 Pragmatics Profile.
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When compared with expected normative values, children with DLI had significantly poorer core language scores (P = .001). Although the mean vocabulary score (PPVT-3) was lower than expected on the basis of normative values, the distribution of scores did not differ significantly from normative data (P = .11). By contrast, the language scores of controls were significantly higher than would be expected on the basis of normative data (P = .04); however, scores on the PPVT-3 did not differ significantly from normative data.
The distributions of nonverbal cognitive testing scores and the results for individual children are shown in Fig 2 and Table 3, respectively. Children with DLI had significantly lower WISC-IV PRI scores than did controls (DLI: 99.5 ± 15.5; controls: 113.5 ± 11.9; P = .009). When compared with controls, children with DLI showed significantly poorer performance on the matrix-reasoning (DLI: 9.1 ± 3.2; controls: 12.7 ± 1.9; P = .002) and block-design (DLI: 9.3 ± 3.5; controls: 11.9 ± 2.6; P = .05) subtests of the WISC-IV. However, the total PRI scores and subtest scores of children with DLI did not differ significantly from published normative values. By contrast, control children had significantly higher total PRI scores (P = .01) and higher scores on the picture-concepts subtests than would be expected on the basis of normative data. Children with DLI showed significantly greater discrepancies between nonverbal cognitive and core language scores than controls (WISC-IV [PRI]/CELF-4 [core language score]—DLI: 17.8 ± 17.8; controls: 1.2 ± 12.7; P = .02) (see Fig 3).
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The distribution of children's scores on the M-ABC is shown in Fig 4, and results for individual children are shown in Table 3. One child in the DLI group did not complete assessment with the M-ABC. The mean impairment score for children with DLI was 14.1 ± 5.3 and for controls was 3.9 ± 5.2 (P = .001). When percentile ranks were considered, the mean M-ABC percentile for children with DLI was 12.7 ± 16.7, whereas for controls the mean was 66.1 ± 30.6. Using an M-ABC score below the 15th percentile to identify impairment, 70% of children with DLI met criteria for motor impairment, whereas 8% of controls met this criteria (P = .003). More than half of the children (6 of 10) with DLI had M-ABC scores that fell below the 5th percentile.
| DISCUSSION |
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Despite a history of delayed language development and ongoing language difficulties in all children with DLI, not all children had abnormal results on the core language tests of the CELF-4. The mean language score fell 1.25 SDs below the expected population mean on the basis of normative data. This corresponds to the 10th percentile cutoff that has been suggested to correlate well with the clinical identification of meaningful language impairment.26 However, several children had core language scores that fell within the reference range, just short of the population mean. Consistent with the clinical diagnosis of DLI, children with DLI showed much greater degrees of discrepancy between language and nonverbal cognitive scores than control children. For a number of children, this discrepancy was the major marker of language impairment.
The PPVT-3 (a test of receptive vocabulary) proved to be a less sensitive instrument for the identification of language impairment than the CELF-4. This is consistent with previous studies that have shown that tests of receptive vocabulary are less sensitive in identifying children with language impairment than more broadly based tests of language.9 Although the PPVT-3 is a relatively easy instrument to administer, it is likely to miss problems with syntax and word structure that are commonly seen in children with DLI.
The range of language findings in this group of children illustrates the heterogeneity of language disorders in school-aged children. The profile of the language abilities of children with DLI has been shown to evolve.27 As children age, earlier problems in areas such as phonology and morphosyntax may improve; however, more detailed testing may show problems with higher-level language (eg, understanding of humor and idioms28). The core language subtests of the CELF-4 (which test receptive and expressive morphosyntax, verbal memory, and verbal working memory and the relationship between words) are insufficient to identify problems with these higher-level language functions. Tests of reading potentially would have an increased sensitivity for detecting language disorders; however, given considerable variability in classroom exposure to reading between families and school environments, we decided not to include these tests.
In this study, articulation was assessed by using the GFTA-2.19 Most of the control children were performing at a ceiling level on this test. Although children with DLI did more poorly than controls, most did not have major problems with their production of individual speech sounds. The sounds-in-words section of the GFTA-2 only examines a child's ability to produce individual speech sounds. A more demanding test of verbal sequencing may be required to demonstrate oromotor language impairments in these children.
For children with DLI, the mean PRI (99.5) was on the 50th percentile on the basis of normative data. The PRI scores of children with DLI had an almost normal distribution and ranged from the 5th percentile to the 90th percentile. This finding is not surprising and is consistent with our recruitment criteria (language impairment in the context of normal nonverbal intelligence). Although the scores of children with DLI were significantly poorer than those of the control population, there was evidence to suggest that the control children were "supernormal."
Motor impairment proved to be a common comorbidity in this group of children. The identification of a group of children on the basis of language impairment also identified a group of children with considerable motor impairment. When the mean percentiles were plotted for the domains tested (Fig 5), levels of motor performance were commensurate with language performance in children with DLI; however, these were unrelated to nonverbal cognitive function. This lends further support to the hypothesis that common biological factors critical to language and motor function (but of relatively less importance for nonverbal cognition) may be the etiologic factors in DLI.14 The number of children enrolled in this study was relatively small; thus, further exploration of the correlation between language, nonverbal cognitive, and motor variables was not possible.
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The relatively high frequency of motor impairments in this cohort is consistent with previous research in this area.4,6,11,12,29 Motor impairment has important consequences for a child's elementary school academic performance (writing, drawing, and coloring) and for a child's ability to participate in sporting and playground activities. Children with language disorders are frequently relatively socially isolated. Motor competence is important for the participation in normal playground activities (eg, ball games, running, and climbing). Children with motor impairments have been found to have lower self-worth and higher levels of anxiety than controls.30 As such, it is possible that comorbid motor impairments exacerbate the social isolation commonly seen in children with DLI. Although the effectiveness of therapy for motor impairment in DLI is uncertain, there is some evidence that treatment can lead to improvements in a child's motor skills.11 Moreover, knowledge of potential motor impairments in children with DLI may lead to targeted surveillance of areas that may cause potential difficulties (ie, pen grip) and guidance regarding physical activities that present fewer motor challenges.
| CONCLUSIONS |
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This study illustrates the considerable clinical heterogeneity seen in school-aged children with DLI. Although motor impairment proved to be a common and significant comorbidity, it was by no means universal. The range of developmental impairments seen in this group of children suggests that the care of children with DLI would be enhanced by multidisciplinary developmental surveillance (eg, speech/language, educational psychology, and occupational therapy). However, given the relatively low number of children with DLI assessed, there is need for additional studies with larger sample sizes to determine if these findings are generally applicable to children with DLI.
| ACKNOWLEDGMENTS |
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Dr Webster was a research fellow supported by the Montreal Children's Hospital Research Institute and the John Yu Scholarship (Children's Hospital at Westmead, Sydney, Australia). Dr Shevell is a chercheur boursier clinicien (clinical research scholar) of the Fonds de Recherche en Santé du Québec. Dr Shevell is also grateful for the support of the Montreal Children's Hospital Foundation. This project was supported by the Réseau Provincial de Recherche en Adaptation-Réadaptation.
We acknowledge the contributions of Nancy Marget, Cynthia Pearlman, Rina Birnbaum, Nicholas Hall, and Lisa Steinbach to this study.
| FOOTNOTES |
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Accepted May 30, 2006.
Address correspondence to Michael I. Shevell, MD, CM, FRCPC, Montreal Children's Hospital, 2300 Tupper St, Room A-514, Montreal, Quebec, Canada H3H 1P3. E-mail: michael.shevell{at}muhc.mcgill.ca
The authors have indicated they have no financial relationships relevant to this article to disclose.
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