Effectiveness of early intervention for children with Down syndrome
01 Jan 1996-Mental Retardation and Developmental Disabilities Research Reviews (John Wiley & Sons, Inc.)-Vol. 2, Iss: 2, pp 96-101
TL;DR: Positive changes were seen in the development of children who were exposed to early intervention programs, and children with Down syndrome and their families are likely to benefit from early intervention.
Abstract: This article reviews studies evaluating the effectiveness of early intervention for children with Down syndrome. Evaluation of early intervention programs is difficult and challenging, given the wide variety of experimental designs and the limitations of research studies. Overall, however, positive changes were seen in the development of children who were exposed to early intervention programs. Children with Down syndrome and their families are likely to benefit from early intervention. © 1996 Wiley-Liss, Inc.
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21 Nov 2008
TL;DR: Early childhood assessment can make crucial contributions to the improvement of children's well-being, but only if they are well designed, implemented effectively, developed in the context of systematic planning, and are interpreted and used appropriately.
Abstract: The assessment of young children's development and learning has recently taken on new importance. Private and government organizations are developing programs to enhance the school readiness of all young children, especially children from economically disadvantaged homes and communities and children with special needs. Well-planned and effective assessment can inform teaching and program improvement, and contribute to better outcomes for children. This book affirms that assessments can make crucial contributions to the improvement of children's well-being, but only if they are well designed, implemented effectively, developed in the context of systematic planning, and are interpreted and used appropriately. Otherwise, assessment of children and programs can have negative consequences for both. The value of assessments therefore requires fundamental attention to their purpose and the design of the larger systems in which they are used. Early Childhood Assessment addresses these issues by identifying the important outcomes for children from birth to age 5 and the quality and purposes of different techniques and instruments for developmental assessments.
309 citations
TL;DR: Analyses of narrative language sample by age sub-group showed no evidence of a critical period for language development ending at adolescence, nor of a "syntactic ceiling" at MLUs corresponding to simple sentences for the Down syndrome group.
Abstract: Hypotheses that children and adolescents with Down syndrome show (a) a specific expressive language impairment, (b) a "critical period" for language acquisition, (c) a "simple sentence syntactic ceiling" in production, and (d) deficit in grammatical morphology were investigated cross-sectionally. Conversational and narrative language samples from 47 children and adolescents with Down syndrome (Trisomy 21), aged 5 to 20 years, were compared to those from 47 control children aged 2 to 6 years matched statistically for nonverbal mental age. Children with Down syndrome appear to have a specific language impairment, compared to control children, in number of different words and total words (in the first 50 utterances) and in mean length of utterance (MLU). Total utterance attempts per minute were more frequent in the Down syndrome group. Narrative samples contained more word tokens, more word types, and longer MLU than conversation samples, for both groups. Intelligibility of narratives was significantly poorer for the Down syndrome group than controls. Analyses of narrative language sample by age sub-group showed no evidence of a critical period for language development ending at adolescence, nor of a "syntactic ceiling" at MLUs corresponding to simple sentences for the Down syndrome group. Omissions of word tokens and types were more frequent in the older Down syndrome than the younger control sample, matched on MLU.
295 citations
TL;DR: In this paper, the authors suggest eight questions that could serve as a framework for determining the extent to which early intervention has accomplished the goals inherent in a family-centered approach, and make recommendations regarding implementation and future directions.
Abstract: Much of the focus on the relationship between parents and professionals in early intervention has been on the rationale for working with families and the processes by which that rationale should be implemented. Although some discussion has occurred regarding desired outcomes, approaches and strategies by which the attainment of family outcomes could be documented have not been widely discussed or agreed upon by the field. In this article we suggest eight questions that could serve as a framework for determining the extent to which early intervention has accomplished the goals inherent in a family-centered approach. Conceptual issues and methodological considerations associated with documenting these outcomes are presented, and recommendations regarding implementation and future directions are made.
294 citations
TL;DR: There is growing consensus based on the best available evidence that early interventions can exert moderate positive effects, but this literature is limited by substantial methodological weaknesses in most studies.
242 citations
TL;DR: The mGluR theory of fragile X syndrome could not be confirmed nor the ability of the methylation state of the FMR1 promoter to predict mavoglurant efficacy, and preclinical results suggest that future clinical trials might profitably explore initiating treatment in a younger population with longer treatment duration and longer placebo run-ins.
Abstract: Fragile X syndrome (FXS), the most common cause of inherited intellectual disability and autistic spectrum disorder, is typically caused by transcriptional silencing of the X-linked FMR1 gene. Work in animal models has described altered synaptic plasticity, a result of the up-regulation of metabotropic glutamate receptor 5 (mGluR5)-mediated signaling, as a putative downstream effect. Post hoc analysis of a randomized, placebo-controlled, crossover phase 2 trial suggested that the selective mGluR5 antagonist mavoglurant improved behavioral symptoms in FXS patients with completely methylated FMR1 genes. We present the results of two phase 2b, multicenter, randomized, double-blind, placebo-controlled, parallel-group studies of mavoglurant in FXS, designed to confirm this result in adults (n = 175, aged 18 to 45 years) and adolescents (n = 139, aged 12 to 17 years). In both trials, participants were stratified by methylation status and randomized to receive mavoglurant (25, 50, or 100 mg twice daily) or placebo over 12 weeks. Neither of the studies achieved the primary efficacy end point of improvement on behavioral symptoms measured by the Aberrant Behavior Checklist-Community Edition using the FXS-specific algorithm (ABC-C(FX)) after 12 weeks of treatment with mavoglurant. The safety and tolerability profile of mavoglurant was as previously described, with few adverse events. Therefore, under the conditions of our study, we could not confirm the mGluR theory of FXS nor the ability of the methylation state of the FMR1 promoter to predict mavoglurant efficacy. Preclinical results suggest that future clinical trials might profitably explore initiating treatment in a younger population with longer treatment duration and longer placebo run-ins and identifying new markers to better assess behavioral and cognitive benefits.
227 citations
Cites result from "Effectiveness of early intervention..."
...This was followed by a similar trial conducted in adolescents (NCT01357239), since preclinical evidence, as well as outcomes from early intervention studies in other genetic disorders with impaired neurocognitive development, indicated that the younger the patient, the greater the potential benefit of the therapeutic intervention (17)....
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References
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TL;DR: The presence of the monosynaptic reflex during platform perturbations at normal latencies suggests that balance problems in children with Down syndrome do not result from hypotonia, but rather result from defects within higher level postural mechanisms.
Abstract: We examined the development of neural control processes underlying stance balance in both developmentally normal children and children with Down syndrome to test the hypothesis that motor deficiencies in children with Down syndrome are associated with deficits within the automatic postural control system. We compared children with Down syndrome and developmentally normal children in two age groups (1-3 and 4-6 years) by using displacements of a platform and measuring electromyograms from leg muscles. The automatic muscle response pattern in both normal children and children with Down syndrome were directionally specific, although the pattern were more variable than in adults. Responses in children with Down syndrome showed no adaptive attenuation to changing task conditions. Onset latencies of responses in children with Down syndrome were significantly slower than in normal children. Presence of the monosynaptic reflex during platform perturbations at normal latencies suggests that balance problems in children with Down syndrome do not result from hypotonia, which researchers have defined as decreased segmental motoneuron pool excitability and pathology of stretch reflex mechanisms, but rather result from defects within higher level postural mechanisms.
280 citations
TL;DR: A cohort of children with Down's syndrome, and their families, have been seen at intervals from six weeks old until 21 yrs when one mother withdrew her son, when no losses were reported apart from those caused by death.
Abstract: A cohort of children with Down's syndrome, and their families, have been seen at intervals from six weeks old. Some follow-up data to 21 yrs are reported here. Until 21 yrs when one mother withdrew her son there have been no losses to the study apart from those caused by death. Mean IQs rose slightly from 11 to 21; scores on language and academic tests favoured the home-reared, females, and middle-class young people even when IQ was allowed for. Some of the effects on the family of having a handicapped member were explored; although adverse effects are fewer than might have been expected many parents, especially mothers, carry significant burdens.
211 citations
TL;DR: Most studies describing early intervention for biologically impaired infants and young children failed to meet common criteria for scientific research, such as specification of inclusion criteria, documentation of reliability, random assignment, and/or the use of control/contrast groups.
Abstract: A review was made of 27 studies describing early intervention for biologically impaired infants and young children. Although every study provided some type of documentation of outcome, most studies failed to meet common criteria for scientific research, such as specification of inclusion criteria, documentation of reliability, random assignment, and/or the use of control/contrast groups. Studies were grouped into four classifications on the basis of experimental design as follows: retrospective, prospective-no control group, prospective-control/contrast group, and prospective-random group assignment. A comparative analysis was made of the effectiveness of findings on the basis of statistical evidence and clinical support. Statistical procedures were used in 59% of the studies and statistical support for the effectiveness of early intervention was reported in 48% of the studies. Effectiveness on the basis of subjective, clinical conclusions was reported in 93% of the studies. Implications of the discrepancy between effectiveness based on statistical evidence and clinical conclusions are discussed.
206 citations
TL;DR: It is hypothesised that Down's syndrome is associated with specific difficulties in using long term motor programmes and that mongol children may therefore be dependent on simple feedback processes to perform motor tasks.
Abstract: SUMMARY
Children with Down's syndrome (Mongolism) were compared with severely subnormal autistic children and with normal children on two simple motor tasks: pursuit rotor tracking and finger tapping. Although the groups were matched on initial tracking performance, Down's syndrome children failed to show any improvement after a 5 min rest, while both comparison groups showed a very marked improvement. Furthermore, in the finger tapping task, Down's syndrome children were abnormally slow compared to the other groups. Neither the level of mental development nor the degree of general mental retardation can account for these deficits. It is hypothesised that Down's syndrome is associated with specific difficulties in using long term motor programmes and that mongol children may therefore be dependent on simple feedback processes to perform motor tasks.
187 citations
01 Jan 1978
TL;DR: Sroufe et al. as mentioned in this paper put many of the outstanding questions in the field of affective development within a unified framework that can be described as an "organizational perspective" that can trace the unfolding of various affect systems and their integration with cognitive and social development.
Abstract: Many of the outstanding questions in the field of affective development may be put within a unified framework that can be described as an “organizational perspective” (Sroufe, in press; Sroufe & Mitchell, in press). For example, the question of age of onset of various emotions becomes subsumed under the objective of tracing the unfolding of various affect systems and their integration with cognitive and social development. The question is not whether the 3-week-old exhibits fear but rather what are the circumstances for eliciting negative reactions at this age and what is the developmental course of both the reactions and the changing conditions necessary and sufficient for eliciting them. How is earlier distress related to what might later be called wariness and later still fear (whatever terms are actually used)? What does the unfolding of the affect systems tell us about cognitive development, and how do maturational and cognitive changes impact upon the expression of affect?
142 citations