The validity of distinguishing children with language and learning disabilities according to discrepancies with IQ: introduction to the special series.
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Achievement↗
Biomedical subjects
J M Fletcher
Publications and source records attributed to J M Fletcher.
The validity of discrepancy-based definitions of reading disabilities.
This study addressed the validity of distinguishing children with reading disabilities according to the presence or absence of discrepancies between intelligence test scores and academic achievement. Three definitions of reading disability were used to provide criteria for five groups of children who (a) met a discrepancy-based definition uncorrected for the correlation of IQ and achievement; (b) met a discrepancy-based definition correcting for the correlation of IQ and achievement; (c) met a low achievement definition with no IQ discrepancy; (d) met criteria a and b; and (e) met none of the criteria and had no reading disability. Comparison of these five groups on a set of 10 neuropsychological tests corrected for correlations with IQ showed that group differences were small and accounted for little of the variability among groups. These results question the validity of segregating children with reading deficiencies according to discrepancies with IQ scores.
Achievement↗
Discrepancy compared to low achievement definitions of reading disability: results from the Connecticut Longitudinal Study.
We used data derived from a survey sample, the Connecticut Longitudinal Study (CLS), to compare two commonly employed definitions of reading disability: a discrepancy-based model (D) and a low reading achievement model (L). We identified children satisfying each definition in second grade and compared the groups retrospectively in kindergarten and prospectively in fifth grade using parent-based, teacher-based, and child-based measures. Our findings suggest more similarities than differences between the reading disabled groups. The most salient differences were those related to ability and seem inherent in the definitions of the groups: Children identified as D have significantly higher verbal, performance, and full scale IQ scores than those identified as L. These findings suggest that both groups of children with reading disability, that is, those defined by either D or L, should be considered eligible for special education services.
Achievement↗
The behavioral sciences and issues in child neurology and developmental pediatrics.
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Behavioral Sciences↗
Submicroscopic deletions at the WAGR locus, revealed by nonradioactive in situ hybridization.
Fluorescence in situ hybridization (FISH) with biotin-labeled probes mapping to 11p13 has been used for the molecular analysis of deletions of the WAGR (Wilms tumor, aniridia, genitourinary abnormalities, and mental retardation) locus. We have detected a submicroscopic 11p13 deletion in a child with inherited aniridia who subsequently presented with Wilms tumor in a horseshoe kidney, only revealed at surgery. The mother, who has aniridia, was also found to carry a deletion including both the aniridia candidate gene (AN2) and the Wilms tumor predisposition gene (WT1). This is therefore a rare case of an inherited WAGR deletion. Wilms tumor has so far only been associated with sporadic de novo aniridia cases. We have shown that a cosmid probe for a candidate aniridia gene, homologous to the mouse Pax-6 gene, is deleted in cell lines from aniridia patients with previously characterized deletions at 11p13, while another cosmid marker mapping between two aniridia-associated translocation breakpoints (and hence a second candidate marker) is present on both chromosomes. These results support the Pax-6 homologue as a strong candidate for the AN2 gene. FISH with cosmid probes has proved to be a fast and reliable technique for the molecular analysis of deletions. It can be used with limited amounts of material and has strong potential for clinical applications.
Adult↗
Neurobehavioral outcomes in diseases of childhood. Individual change models for pediatric human immunodeficiency viruses.
The growing incidence of human immunodeficiency virus-1 (HIV-1) and acquired immunodeficiency syndrome (AIDS) in children is a major public health problem. Current research emphasizes treatments for ameliorating deleterious effects on the child's neurological and behavioral development. This article outlines approaches to the assessment of individual change that may provide alternatives to more traditional approaches to the assessment of neurobehavioral outcomes in children with chronic diseases. These approaches provide more precise conceptualizations of changes that lead directly to statistical designs and measurement strategies for assessing effects of HIV-1 and AIDS on development. Such assessments can be superimposed on current clinical trial methodologies to evaluate the efficacy of pharmacological and behavioral interventions designed to improve quality of life in HIV-1 infected children.
Acquired Immunodeficiency Syndrome↗
Analysis of change: modeling individual growth.
Research on change is complicated by problems of measurement and analysis stemming from a conceptualization of change as a series of accumulating increments and decrements. In contrast, individual growth curves depict change as a continuous process underlying individual performance. These two perspectives are reviewed, and some problems with the use of difference scores in the study of change are clarified. Traditional methods are contrasted with growth curve analysis for the purposes of measuring change and studying its correlates. An illustrative example of the use of growth curves is provided from research on recovery of cognitive function following pediatric closed head injury.
Adolescent↗
Cognitive and motor abilities in preschool hydrocephalics.
The neuropsychological performance of three groups of preschool children was evaluated: (a) one with hydrocephalus associated with myelomeningocele; (b) one with hydrocephalus associated with intraventricular hemorrhage and very low birth weight; and (c) a nonhydrocephalic normal comparison group. Multivariate profile analysis revealed lower levels of performance on measures of verbal and nonverbal cognitive skills for both groups of hydrocephalic children relative to normals. Comparison of group profiles on tasks requiring figure copying as opposed to figure matching and analysis of specific gross and fine motor skills revealed that both hydrocephalic groups had impaired visual-motor integration in the presence of average visual perceptual matching. In addition, different patterns of motor skill deficits were found for each hydrocephalic group. The results of this study suggest that decreased visual-motor integration and etiology-specific motor deficits are major sequelae of these forms of hydrocephalus in the preschool years.
Cerebral Hemorrhage↗
Left unilateral neglect in a preschool child.
The authors present a case of transient left hemispatial inattention following traumatic brain injury in a three-year-old child. Computed tomographic scan revealed a small right frontal and a right temporo-parietal contusion. Stimuli were developed to quantify the response difference between hemifields in this pre-literate child. Hemispatial inattention showed resolution at 19 days post-trauma and had completely resolved six months later. These findings suggest that right hemisphere systems subserve visually guided attention before six years of age.
Attention↗
Methodological issues in the classification of attention-related disorders.
The classification and identification of children with attention deficit-hyperactivity disorder and related disorders involving learning and behavior are only rudimentarily developed. A major problem is the substantial overlap among children with attention, learning, and behavioral problems. The science of classification provides conceptual and methodological approaches addressing these problems. For successful classification of these children, major issues include (a) the need to provide explicit studies of identification criteria, (b) the need for systematic sampling strategies, (c) development of hypothetical classifications, and (d) systematic assessment of reliability and validity of hypothetical classifications. With the methodological advances provided by classification-oriented research, investigators and practitioners may be able to address issues involving definition and identification of children with attention and related disorders.
Attention Deficit Disorder with Hyperactivity↗
Absence of synergistic effects of CNS treatments on neuropsychologic test performance among children.
Three hypotheses are proposed to account for neurobehavioral impairments following treatment with cranial radiation therapy (CRT) and intrathecal (IT) chemotherapy: CNS treatments exert a synergistic effect (A x B), an additive effect (A + B), or a single-agent effect (A or B). Eighty-five long-term survivors of non-CNS cancers aged 6 to 16 years were classified into groups on the basis of CNS treatments: CRT-IT (n = 25), CRT-No IT (n = 11), No CRT-IT (n = 24), and No CRT-No IT (n = 25). Study I findings did not provide support for synergistic mechanisms; nonorthogonal analysis of variance showed interaction effects (CRT x IT) restricted to tactile-perceptual speed. However, main effects were significant for a single agent (CRT) across a wide range of measures. General intelligence, academic achievement, verbal knowledge and reasoning, and perceptual-motor abilities were found to be significantly lower among CRT-treated groups. Study II findings provided additional support for the role of CRT; Pearson correlations within the CRT-No IT group indicated significant negative associations between CRT dose estimates for cortical regions and perceptual-motor abilities.
Adolescent↗
SV40-mediated tumor selection and chromosome transfer to enrich for cystic fibrosis region.
The somatic cell hybrid C121, with chromosome 7 as its sole human component, arose when mouse macrophages SV40 genomes are integrated at 7q31-7q35. We show that hybrids with a reduced chromosome 7 component, but which retain markers linked to the cystic fibrosis locus, can be generated by direct in vivo tumor selection or following chromosome-mediated gene transfer and SV40-mediated cellular transformation. Our methods for chromosome fragmentation and fine-structure mapping can now be applied to the substantial number of SV40-transformed human cell lines, with independent chromosomal integration sites, already available. Our results also suggest that expression of human epidermal growth factor receptor augments the tumorigenic potential of the SV40-transformed C121 hybrid.
Animals↗
Behavioral changes after closed head injury in children.
This study provides a longitudinal follow-up of the behavioral adjustment of 45 children with mild, moderate, and severe closed head injuries. Two measures of behavioral adjustment, the Child Behavior Checklist (CBCL) and the Vineland Adaptive Behavior Scales (VABS), were obtained from a parent at the time of injury and at 6 and 12 months postinjury. The severely injured children obtained significantly poorer VABS scores than children with mild and moderate injuries over the year-long follow-up. In addition, on the CBCL, severely injured children had more school problems and engaged in fewer social activities than mild and moderately injured children. These results show that severe head injury in children was associated with declines in adaptive functioning, whereas scores for children with mild and moderate injuries did not differ, nor did they deviate from average levels at any follow-up interval.
Age Factors↗
Role for the Wilms tumor gene in genital development?
Detailed molecular definition of the WAGR region at chromosome 11p13 has been achieved by chromosome breakpoint analysis and long-range restriction mapping. Here we describe the molecular detection of a cytogenetically invisible 1-megabase deletion in an individual with aniridia, cryptorchidism, and hypospadias but no Wilms tumor (WT). The region of overlap between this deletion and one associated with WT and similar genital anomalies but no aniridia covers a region of 350-400 kilobases, which is coincident with the extent of homozygous deletion detected in tumor tissue from a sporadic WT. A candidate WT gene located within this region has recently been isolated, suggesting nonpenetrance for tumor expression in the first individual. The inclusion within the overlap region of a gene for WT predisposition and a gene for the best-documented WT-associated genitourinary malformations leads us to suggest that both of these anomalies result from a loss-of-function mutation at the same locus. This in turn implies that the WT gene exerts pleiotropic effect on both kidney and genitourinary development, a possibility supported by the observed expression pattern of the WT candidate gene in developing kidney and gonads.
Aniridia↗
Verbal and nonverbal communication of events in learning-disability subtypes.
This study compared a group of nondisabled children (ND) with groups of learning-disabled children who were primarily impaired in reading and arithmetic skills (Reading-Arithmetic Disabled; RAD) and arithmetic but not reading (Arithmetic Disabled; AD) on a set of tasks involving comprehension and production of verbally and nonverbally presented events. Children viewed videotaped scenarios presented in verbal (narrative) and nonverbal (puppet actors) formats and were asked to describe or enact with puppets the events depicted in the stories. Rourke (1978, 1982) has shown that RAD children have problems with verbal skills, whereas AD children have problems with nonverbal skills. Consequently, it was hypothesized that children's performance in comprehending and reproducing stories would be related to the type of learning disability. Results showed that RAD children made more errors than AD children with verbal presentations and describe-responses, whereas AD children made more errors than RAD children with nonverbal presentations and enact-responses. In addition, learning disabled children were more likely than controls to misinterpret affect and motivation depicted in the stories. These results show that learning disabled children have problems with social communication skills, but that the nature of these problems varies with the type of learning disability.
Affect↗
Isolation and use of chromosome 1 probes for linkage studies on Charcot-Marie-Tooth disease.
Nine probes were isolated from a human chromosome 1 enriched library and mapped to regions of chromosome 1 using somatic cell hybrid lines. One clone, LR67, which mapped to 1q12----q23 detected a BglI RFLP. This probe, as well as 4 other known chromosome 1 markers, alpha-spectrin, Factor XIIIB, DR10 and DR78, were used for linkage studies in 15 Charcot-Marie-Tooth disease (CMT1) families. Close linkage of CMT1 to any of the 5 markers was not indicated. Total lod scores excluded linkage of CMT1 to LR67 and to DR10 at 5 cM or less, to DR78 at 10 cM or less, alpha-spectrin at 15 cM or less and Factor XIIIB at 20 cM or less. Possible linkage, however, was shown between LR67 and CMT1 at a distance of 30 cM. Also linkage at a distance of 5 cM was detected between this probe and alpha-spectrin.
Charcot-Marie-Tooth Disease↗
The Children's Orientation and Amnesia Test: relationship to severity of acute head injury and to recovery of memory.
The Children's Orientation and Amnesia Test (COAT) was developed to assess cognition serially during the early stage of recovery from traumatic brain injury in children and adolescents. The norms for the COAT, which is composed of 16 items evaluating general orientation, temporal orientation, and memory, were defined from data obtained from 146 children aged 3 to 15 years. In 37 patients with head injuries, the duration of posttraumatic amnesia, as indicated by the number of days COAT scores were in the impaired range, was significantly related to both verbal and nonverbal memory at the baseline and 6 and 12 months after injury. COAT scores were a better predictor of verbal and nonverbal memory performance than the Glasgow Coma Scale score at 6 and 12 months after the injury. This study shows that the COAT has adequate reliability and validity as a measure of the duration of posttraumatic amnesia in children and adolescents.
Acute Disease↗
Human-mouse hybrids carrying fragments of single human chromosomes selected by tumor growth.
Fusion of human EJ bladder carcinoma cells to mouse C127 cells, with direct selection for tumor growth, gave rise to hybrid cells in which the human chromosome complement had been reduced dramatically, while selectively retaining the activated HRAS1 at chromosome band 11p15. A single-component hybrid retaining only part of human chromosome 11 is described in detail. Our results suggest a novel and general approach for investigating the chromosomal basis of neoplastic change and for subchromosomal mapping of and enrichment cloning for the human genome.
Animals↗