77 results on '"Slonims, V"'
Search Results
2. Genome-wide analysis identifies a role for common copy number variants in specific language impairment
- Author
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Simpson N. H., Ceroni F., Reader R. H., Covill L. E., Knight J. C., Hennessy E. R., Bolton P. F., Conti-Ramsden G., O'Hare A., Baird G., Fisher S. E., Newbury D. F., Nudel R., Monaco A. P., Simonoff E., Pickles A., Slonims V., Dworzynski K., Everitt A., Clark A., Watson J., Seckl J., Cowie H., Cohen W., Nasir J., Bishop D. V. M., Simkin Z., Simpson N.H., Ceroni F., Reader R.H., Covill L.E., Knight J.C., Nudel R., Monaco A.P., Simonoff E., Pickles A., Slonims V., Dworzynski K., Everitt A., Clark A., Watson J., Seckl J., Cowie H., Cohen W., Nasir J., Bishop D.V.M., Simkin Z, Hennessy E.R., Bolton P.F., Conti-Ramsden G., O'Hare A., Baird G., Fisher S.E., and Newbury D.F.
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Male ,Language Disorders ,Genome ,DNA Copy Number Variations ,Specific Language Impairment ,Polymorphism, Single Nucleotide ,Article ,Copy Number Variations ,Case-Control Studies ,Humans ,Female ,Genetic Predisposition to Disease ,Aged ,Genome-Wide Association Study ,Language - Abstract
An exploratory genome-wide copy number variant (CNV) study was performed in 127 independent cases with specific language impairment (SLI), their first-degree relatives (385 individuals) and 269 population controls. Language-impaired cases showed an increased CNV burden in terms of the average number of events (11.28 vs 10.01, empirical P=0.003), the total length of CNVs (717 vs 513 Kb, empirical P=0.0001), the average CNV size (63.75 vs 51.6 Kb, empirical P=0.0005) and the number of genes spanned (14.29 vs 10.34, empirical P=0.0007) when compared with population controls, suggesting that CNVs may contribute to SLI risk. A similar trend was observed in first-degree relatives regardless of affection status. The increased burden found in our study was not driven by large or de novo events, which have been described as causative in other neurodevelopmental disorders. Nevertheless, de novo CNVs might be important on a case-by-case basis, as indicated by identification of events affecting relevant genes, such as ACTR2 and CSNK1A1, and small events within known micro-deletion/-duplication syndrome regions, such as chr8p23.1. Pathway analysis of the genes present within the CNVs of the independent cases identified significant overrepresentation of acetylcholine binding, cyclic-nucleotide phosphodiesterase activity and MHC proteins as compared with controls. Taken together, our data suggest that the majority of the risk conferred by CNVs in SLI is via common, inherited events within a 'common disorder-common variant' model. Therefore the risk conferred by CNVs will depend upon the combination of events inherited (both CNVs and SNPs), the genetic background of the individual and the environmental factors.
- Published
- 2014
3. Increased prevalence of sex chromosome aneuploidies in specific language impairment and dyslexia
- Author
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Simpson, N, Addis, L, Brandler, WM, Slonims, V, Clark, A, Watson, J, Scerri, T, Hennessy, E, Bolton, P, Conti-Ramsden, G, Fairfax, B, Knight, J, Stein, J, Talcott, J, O'Hare, A, Baird, G, Paracchini, S, Fisher, SE, Newbury, D, Nudel, R, Monaco, A, Simonoff, E, Pickles, A, Everitt, A, and Seckl, J
- Abstract
Aim: Sex chromosome aneuploidies increase the risk of spoken or written language disorders but individuals with specific language impairment (SLI) or dyslexia do not routinely undergo cytogenetic analysis. We assess the frequency of sex chromosome aneuploidies in individuals with language impairment or dyslexia. Method: Genome-wide single nucleotide polymorphism genotyping was performed in three sample sets: a clinical cohort of individuals with speech and language deficits (87 probands: 61 males, 26 females; age range 4 to 23 years), a replication cohort of individuals with SLI, from both clinical and epidemiological samples (209 probands: 139 males, 70 females; age range 4 to 17 years), and a set of individuals with dyslexia (314 probands: 224 males, 90 females; age range 7 to 18 years). Results: In the clinical language-impaired cohort, three abnormal karyotypic results were identified in probands (proband yield 3.4%). In the SLI replication cohort, six abnormalities were identified providing a consistent proband yield (2.9%). In the sample of individuals with dyslexia, two sex chromosome aneuploidies were found giving a lower proband yield of 0.6%. In total, two XYY, four XXY (Klinefelter syndrome), three XXX, one XO (Turner syndrome), and one unresolved karyotype were identified. Interpretation: The frequency of sex chromosome aneuploidies within each of the three cohorts was increased over the expected population frequency (approximately 0.25%) suggesting that genetic testing may prove worthwhile for individuals with language and literacy problems and normal non-verbal IQ. Early detection of these aneuploidies can provide information and direct the appropriate management for individuals. © 2013 The Authors. Developmental Medicine and Child Neurology published by John Wiley and Sons Ltd on behalf of Mac Keith Press.
- Published
- 2016
4. Increased prevalence of sex chromosome aneuploidies in specific language impairment and dyslexia
- Author
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Simpson, NH, Addis, L, Brandler, WM, Slonims, V, Clark, A, Watson, J, Scerri, TS, Hennessy, ER, Bolton, PF, Conti-Ramsden, G, Fairfax, BP, Knight, JC, Stein, J, Talcott, JB, O'Hare, A, Baird, G, Paracchini, S, Fisher, SE, and Newbury, DF
- Abstract
AIM: Sex chromosome aneuploidies increase the risk of spoken or written language disorders but individuals with specific language impairment (SLI) or dyslexia do not routinely undergo cytogenetic analysis. We assess the frequency of sex chromosome aneuploidies in individuals with language impairment or dyslexia. METHOD: Genome-wide single nucleotide polymorphism genotyping was performed in three sample sets: a clinical cohort of individuals with speech and language deficits (87 probands: 61 males, 26 females; age range 4 to 23 years), a replication cohort of individuals with SLI, from both clinical and epidemiological samples (209 probands: 139 males, 70 females; age range 4 to 17 years), and a set of individuals with dyslexia (314 probands: 224 males, 90 females; age range 7 to 18 years). RESULTS: In the clinical language-impaired cohort, three abnormal karyotypic results were identified in probands (proband yield 3.4%). In the SLI replication cohort, six abnormalities were identified providing a consistent proband yield (2.9%). In the sample of individuals with dyslexia, two sex chromosome aneuploidies were found giving a lower proband yield of 0.6%. In total, two XYY, four XXY (Klinefelter syndrome), three XXX, one XO (Turner syndrome), and one unresolved karyotype were identified. INTERPRETATION: The frequency of sex chromosome aneuploidies within each of the three cohorts was increased over the expected population frequency (approximately 0.25%) suggesting that genetic testing may prove worthwhile for individuals with language and literacy problems and normal non-verbal IQ. Early detection of these aneuploidies can provide information and direct the appropriate management for individuals.
- Published
- 2016
5. Genome-wide analysis identifies a role for common copy number variants in specific language impairment
- Author
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Simpson, Nuala H, Ceroni, Fabiola, Reader, Rose H, Covill, Laura E, Knight, Julian C, Nudel, R, Monaco, A P, Simonoff, E, Bolton, P F, Pickles, A, Slonims, V, Dworzynski, K, Everitt, A, Clark, A, Watson, J, Seckl, J, Cowie, H, Cohen, W, Nasir, Jamal, Bishop, D V M, Simkin, Z, Hennessy, Elizabeth R, Bolton, Patrick F, Conti-Ramsden, Gina, O'Hare, Anne, Baird, Gillian, Fisher, Simon E, and Newbury, Dianne F
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RZ ,A100 ,QH426 - Abstract
An exploratory genome-wide copy number variant (CNV) study was performed in 127 independent cases with specific language impairment (SLI), their first-degree relatives (385 individuals) and 269 population controls. Language-impaired cases showed an increased CNV burden in terms of the average number of events (11.28 vs 10.01, empirical P=0.003), the total length of CNVs (717 vs 513 Kb, empirical P=0.0001), the average CNV size (63.75 vs 51.6 Kb, empirical P=0.0005) and the number of genes spanned (14.29 vs 10.34, empirical P=0.0007) when compared with population controls, suggesting that CNVs may contribute to SLI risk. A similar trend was observed in first-degree relatives regardless of affection status. The increased burden found in our study was not driven by large or de novo events, which have been described as causative in other neurodevelopmental disorders. Nevertheless, de novo CNVs might be important on a case-by-case basis, as indicated by identification of events affecting relevant genes, such as ACTR2 and CSNK1A1, and small events within known micro-deletion/-duplication syndrome regions, such as chr8p23.1. Pathway analysis of the genes present within the CNVs of the independent cases identified significant overrepresentation of acetylcholine binding, cyclic-nucleotide phosphodiesterase activity and MHC proteins as compared with controls. Taken together, our data suggest that the majority of the risk conferred by CNVs in SLI is via common, inherited events within a 'common disorder-common variant' model. Therefore the risk conferred by CNVs will depend upon the combination of events inherited (both CNVs and SNPs), the genetic background of the individual and the environmental factors.
- Published
- 2015
6. Lack of replication for the myosin-18B association with mathematical ability in independent cohorts
- Author
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Pettigrew, K. A., Fajutrao Valles, S. F., Moll, K., Northstone, K., Ring, S., Pennell, C., Wang, C., Leavett, R., Hayiou-Thomas, M. E., Thompson, P., Simpson, N. H., Fisher, S. E., Whitehouse, A. J O, Snowling, M. J., Newbury, D. F., Paracchini, S., Nudel, R., Monaco, A. P., Francks, C., Baird, G., Slonims, V., Dworzynski, K., Bolton, P. F., Simonoff, E., O'Hare, A., Seckl, J., Cowie, H., Clark, A., Watson, J., Nasir, J., Cohen, W., Everitt, A., Hennessy, E. R., Shaw, D., Helms, P. J., Simkin, Z., Conti, G., Ramsden, D., Bishop, D. V M, and Pickles, A.
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RM - Abstract
Twin studies indicate that dyscalculia (or mathematical disability) is caused partly by a genetic component, which is yet to be understood at the molecular level. Recently, a coding variant (rs133885) in the myosin-18B gene was shown to be associated with mathematical abilities with a specific effect among children with dyslexia. This association represents one of the most significant genetic associations reported to date for mathematical abilities and the only one reaching genome-wide statistical significance. We conducted a replication study in different cohorts to assess the effect of rs133885 maths-related measures. The study was conducted primarily using the Avon Longitudinal Study of Parents and Children (ALSPAC), (N=3819). We tested additional cohorts including the York Cohort, the Specific Language Impairment Consortium (SLIC) cohort and the Raine Cohort, and stratified them for a definition of dyslexia whenever possible. We did not observe any associations between rs133885 in myosin-18B and mathematical abilities among individuals with dyslexia or in the general population. Our results suggest that the myosin-18B variant is unlikely to be a main factor contributing to mathematical abilities. We could not replicate the association of the myosin-18B gene with mathematical ability.
- Published
- 2015
7. Increased prevalence of sex chromosome aneuploidies in specific language impairment and dyslexia
- Author
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Simpson, NH, Addis, L, Brandler, WM, Slonims, V, Clark, A, Watson, J, Scerri, TS, Hennessy, ER, Bolton, PF, Conti-Ramsden, G, Fairfax, BP, Knight, JC, Stein, J, Talcott, JB, O'Hare, A, Baird, G, Paracchini, S, Fisher, SE, Newbury, DF, Nudel, R, Monaco, AP, Simonoff, E, Pickles, A, Everitt, A, Seckl, J, Cowie, H, Cohen, W, Nasir, J, Bishop, DVM, and Simkin, Z
- Abstract
Aim: Sex chromosome aneuploidies increase the risk of spoken or written language disorders but individuals with specific language impairment (SLI) or dyslexia do not routinely undergo cytogenetic analysis. We assess the frequency of sex chromosome aneuploidies in individuals with language impairment or dyslexia. Method: Genome-wide single nucleotide polymorphism genotyping was performed in three sample sets: a clinical cohort of individuals with speech and language deficits (87 probands: 61 males, 26 females; age range 4 to 23 years), a replication cohort of individuals with SLI, from both clinical and epidemiological samples (209 probands: 139 males, 70 females; age range 4 to 17 years), and a set of individuals with dyslexia (314 probands: 224 males, 90 females; age range 7 to 18 years). Results: In the clinical language-impaired cohort, three abnormal karyotypic results were identified in probands (proband yield 3.4%). In the SLI replication cohort, six abnormalities were identified providing a consistent proband yield (2.9%). In the sample of individuals with dyslexia, two sex chromosome aneuploidies were found giving a lower proband yield of 0.6%. In total, two XYY, four XXY (Klinefelter syndrome), three XXX, one XO (Turner syndrome), and one unresolved karyotype were identified. Interpretation: The frequency of sex chromosome aneuploidies within each of the three cohorts was increased over the expected population frequency (approximately 0.25%) suggesting that genetic testing may prove worthwhile for individuals with language and literacy problems and normal non-verbal IQ. Early detection of these aneuploidies can provide information and direct the appropriate management for individuals. © 2013 The Authors. Developmental Medicine and Child Neurology published by John Wiley and Sons Ltd on behalf of Mac Keith Press.
- Published
- 2014
8. Exome Sequencing in an Admixed Isolated Population Indicates NFXL1 Variants Confer a Risk for Specific Language Impairment
- Author
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Villanueva, Pía, Nudel, Ron, Hoischen, Alexander, Fernández, María Angélica, Simpson, Nuala H., Gilissen, Christian, Reader, Rose H., Jara, Lillian, Echeverry, Maria Magdalena, Francks, Clyde, Baird, Gillian, Conti-Ramsden, Gina, O’Hare, Anne, Bolton, Patrick F., Hennessy, Elizabeth R., Palomino, Hernán, Carvajal-Carmona, Luis, Veltman, Joris A., Cazier, Jean Baptiste, De Barbieri, Zulema, Fisher, Simon E., Newbury, Dianne F., Slonims, V., Clark, A., Watson, J., Simonoff, Emily, Pickles, Andrew, Everitt, A., Seckl, J., Cowie, H., Cohen, W., Nasir, J., Bishop, D. V M, Simkin, Z., and Abrahams, Brett S
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Male ,Cancer Research ,SLI Consortium ,Genome-wide association study ,Specific language impairment ,0302 clinical medicine ,2.1 Biological and endogenous factors ,Exome ,Aetiology ,Child ,Genetics (clinical) ,Exome sequencing ,Pediatric ,Genetics ,0303 health sciences ,education.field_of_study ,High-Throughput Nucleotide Sequencing ,Female ,Mental health ,Research Article ,Neuroinformatics ,lcsh:QH426-470 ,Apraxias ,Population ,Biology ,behavioral disciplines and activities ,03 medical and health sciences ,Clinical Research ,Genetic model ,medicine ,Humans ,Genetic Predisposition to Disease ,Allele ,Preschool ,education ,QH426 ,Molecular Biology ,Genetic Association Studies ,Ecology, Evolution, Behavior and Systematics ,030304 developmental biology ,Neurodevelopmental disorders Donders Center for Medical Neuroscience [Radboudumc 7] ,Human Genome ,Membrane Proteins ,medicine.disease ,Minor allele frequency ,lcsh:Genetics ,RF ,Carrier Proteins ,030217 neurology & neurosurgery ,Developmental Biology ,Founder effect - Abstract
Children affected by Specific Language Impairment (SLI) fail to acquire age appropriate language skills despite adequate intelligence and opportunity. SLI is highly heritable, but the understanding of underlying genetic mechanisms has proved challenging. In this study, we use molecular genetic techniques to investigate an admixed isolated founder population from the Robinson Crusoe Island (Chile), who are affected by a high incidence of SLI, increasing the power to discover contributory genetic factors. We utilize exome sequencing in selected individuals from this population to identify eight coding variants that are of putative significance. We then apply association analyses across the wider population to highlight a single rare coding variant (rs144169475, Minor Allele Frequency of 4.1% in admixed South American populations) in the NFXL1 gene that confers a nonsynonymous change (N150K) and is significantly associated with language impairment in the Robinson Crusoe population (p = 2.04 × 10–4, 8 variants tested). Subsequent sequencing of NFXL1 in 117 UK SLI cases identified four individuals with heterozygous variants predicted to be of functional consequence. We conclude that coding variants within NFXL1 confer an increased risk of SLI within a complex genetic model., Author Summary Children affected by Specific Language Impairment (SLI) have unexpected problems learning to talk and understand language, despite developing normally in all other areas. This disorder runs in families but we do not understand how the genetic contributions work, or which genetic mechanisms might be important. In this paper, we study a Chilean population who are affected by a high incidence of SLI. Such populations may provide increased power to discover contributory genetic factors, under appropriate conditions. We identify a genetic change in the population that causes a change to a protein called NFXL1. This change is usually very rare but is found at a higher frequency than expected in our population, particularly in those people affected by SLI. We then looked at this gene in over 100 individuals from the UK affected by SLI and found four more changes that probably affect the protein. This is a higher number than we would expect by chance. We therefore propose that the NFXL1 gene and the protein it encodes might be important in risk of SLI.
- Published
- 2015
9. Highly significant linkage to the SLI1 locus in an expanded sample of individuals affected by specific language impairment
- Author
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Newbury, DF, Cleak, JD, Banfield, E, Marlow, AJ, Fisher, SE, Monaco, AP, Stott, CM, Merricks, MJ, Goodyer, IM, Bolton, PF, Slonims, V, Baird, G, Everitt, A, Hennessy, E, Main, M, Helms, P, Kindley, AD, Hodson, A, Watson, J, O'Hare, A, Cohen, W, Cowie, H, Steel, J, MacLean, A, Seckl, J, Nasir, J, Bishop, DVM, Simkin, Z, Conti-Ramsden, G, Pickles, A, and Consortium, SLI
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Male ,Reading disability ,Adolescent ,Genotype ,Src Homology 2 Domain-Containing, Transforming Protein 2 ,Genetic Linkage ,Quantitative Trait Loci ,Twins ,Locus (genetics) ,Biology ,Specific language impairment ,Nuclear Family ,Cohort Studies ,03 medical and health sciences ,0302 clinical medicine ,Chromosome 16 ,Genetic linkage ,Chromosome 19 ,Genetics ,medicine ,Diseases in Twins ,Humans ,Computer Simulation ,Genetic Predisposition to Disease ,Language Development Disorders ,Child ,Nuclear family ,Genetics (clinical) ,030304 developmental biology ,Adaptor Proteins, Signal Transducing ,0303 health sciences ,Models, Genetic ,Genome, Human ,Wechsler Adult Intelligence Scale ,Chromosome Mapping ,Proteins ,Articles ,medicine.disease ,Pedigree ,Phenotype ,England ,Shc Signaling Adaptor Proteins ,Child, Preschool ,Female ,Lod Score ,Chromosomes, Human, Pair 19 ,030217 neurology & neurosurgery ,Chromosomes, Human, Pair 16 - Abstract
Specific language impairment (SLI) is defined as an unexplained failure to acquire normal language skills despite adequate intelligence and opportunity. We have reported elsewhere a full-genome scan in 98 nuclear families affected by this disorder, with the use of three quantitative traits of language ability (the expressive and receptive tests of the Clinical Evaluation of Language Fundamentals and a test of nonsense word repetition). This screen implicated two quantitative trait loci, one on chromosome 16q (SLI1) and a second on chromosome 19q (SLI2). However, a second independent genome screen performed by another group, with the use of parametric linkage analyses in extended pedigrees, found little evidence for the involvement of either of these regions in SLI. To investigate these loci further, we have collected a second sample, consisting of 86 families (367 individuals, 174 independent sib pairs), all with probands whose language skills are greater-or-equal, slanted1.5 SD below the mean for their age. Haseman-Elston linkage analysis resulted in a maximum LOD score (MLS) of 2.84 on chromosome 16 and an MLS of 2.31 on chromosome 19, both of which represent significant linkage at the 2% level. Amalgamation of the wave 2 sample with the cohort used for the genome screen generated a total of 184 families (840 individuals, 393 independent sib pairs). Analysis of linkage within this pooled group strengthened the evidence for linkage at SLI1 and yielded a highly significant LOD score (MLS = 7.46, interval empirical P
- Published
- 2004
10. FOXP2 is not a major susceptibility gene for autism or specific language impairment
- Author
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Newbury, D., Bonora, E., Lamb, J., Fisher, S., Lai, C., Baird, G., Jannoun, L., Slonims, V., Stott, C., Merricks, M., Bolton, P., Bailey, A., Monaco, A., and International Molecular Genetic Study of Autism Consortium
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Male ,DNA Mutational Analysis ,Molecular Sequence Data ,Biology ,Specific language impairment ,Polymorphism, Single Nucleotide ,03 medical and health sciences ,0302 clinical medicine ,Speech and language impairment ,Gene Frequency ,Report ,medicine ,Genetics ,Humans ,Heritability of autism ,Language disorder ,Genetics(clinical) ,Genetic Predisposition to Disease ,Amino Acid Sequence ,Genetic Testing ,Autistic Disorder ,Genetics (clinical) ,Alleles ,030304 developmental biology ,0303 health sciences ,Language Disorders ,FOXP2 Gene ,Base Sequence ,FOXP2 ,Forkhead Transcription Factors ,Exons ,medicine.disease ,Introns ,Pedigree ,Developmental disorder ,Repressor Proteins ,Autism ,Female ,030217 neurology & neurosurgery ,Microsatellite Repeats ,Transcription Factors - Abstract
The FOXP2 gene, located on human 7q31 (at the SPCH1 locus), encodes a transcription factor containing a polyglutamine tract and a forkhead domain. FOXP2 is mutated in a severe monogenic form of speech and language impairment, segregating within a single large pedigree, and is also disrupted by a translocation in an isolated case. Several studies of autistic disorder have demonstrated linkage to a similar region of 7q (the AUTS1 locus), leading to the proposal that a single genetic factor on 7q31 contributes to both autism and language disorders. In the present study, we directly evaluate the impact of the FOXP2 gene with regard to both complex language impairments and autism, through use of association and mutation screening analyses. We conclude that coding-region variants in FOXP2 do not underlie the AUTS1 linkage and that the gene is unlikely to play a role in autism or more common forms of language impairment.
- Published
- 2002
11. A genomewide scan identifies two novel loci involved in specific language impairment
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Newbury, D. F., Ishikawa-Brush, Y., Marlow, A. J., Fisher, S. E., Monaco, A. P., Stott, C. M., Merricks, M. J., Goodyer, I. M., Patrick Bolton, Jannoun, L., Slonims, V., Baird, G., Andrew Pickles, Bishop, D. V. M., Conti-Ramsden, G., Helms, P. J., and Sli, Consortium
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Male ,X Chromosome ,Adolescent ,Genotype ,Intelligence ,Twins ,Nuclear Family ,Cohort Studies ,Quantitative Trait, Heritable ,Diseases in Twins ,Humans ,Computer Simulation ,Genetic Predisposition to Disease ,Language Development Disorders ,Child ,Language Tests ,Models, Genetic ,Genome, Human ,Chromosome Mapping ,Articles ,Phenotype ,England ,Child, Preschool ,Female ,Lod Score ,Chromosomes, Human, Pair 19 ,Chromosomes, Human, Pair 16 - Abstract
Approximately 4% of English-speaking children are affected by specific language impairment (SLI), a disorder in the development of language skills despite adequate opportunity and normal intelligence. Several studies have indicated the importance of genetic factors in SLI; a positive family history confers an increased risk of development, and concordance in monozygotic twins consistently exceeds that in dizygotic twins. However, like many behavioral traits, SLI is assumed to be genetically complex, with several loci contributing to the overall risk. We have compiled 98 families drawn from epidemiological and clinical populations, all with probands whose standard language scores fallor =1.5 SD below the mean for their age. Systematic genomewide quantitative-trait-locus analysis of three language-related measures (i.e., the Clinical Evaluation of Language Fundamentals-Revised [CELF-R] receptive and expressive scales and the nonword repetition [NWR] test) yielded two regions, one on chromosome 16 and one on 19, that both had maximum LOD scores of 3.55. Simulations suggest that, of these two multipoint results, the NWR linkage to chromosome 16q is the most significant, with empirical P values reaching 10(-5), under both Haseman-Elston (HE) analysis (LOD score 3.55; P=.00003) and variance-components (VC) analysis (LOD score 2.57; P=.00008). Single-point analyses provided further support for involvement of this locus, with three markers, under the peak of linkage, yielding LOD scores1.9. The 19q locus was linked to the CELF-R expressive-language score and exceeds the threshold for suggestive linkage under all types of analysis performed-multipoint HE analysis (LOD score 3.55; empirical P=.00004) and VC (LOD score 2.84; empirical P=.00027) and single-point HE analysis (LOD score 2.49) and VC (LOD score 2.22). Furthermore, both the clinical and epidemiological samples showed independent evidence of linkage on both chromosome 16q and chromosome 19q, indicating that these may represent universally important loci in SLI and, thus, general risk factors for language impairment.
- Published
- 2002
12. Regression in individuals with Rett Syndrome
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Tony Charman, Cass, H., Owen, L., Wigram, T., Slonims, V., Weeks, L., Wisbeach, A., and Reilly, S.
- Published
- 2002
13. Findings from a multidisciplinary clinical case series of females with Rett syndrome.
- Author
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Cass H, Reilly S, Owen L, Wisbeach A, Charman T, Weekes L, Slonims V, Wigram T, Cass, Hilary, Reilly, Sheena, Owen, Lucy, Wisbeach, Alison, Weekes, Lyn, Slonims, Vicky, Wigram, Tony, and Charman, Tony
- Abstract
Systematic data from a multidisciplinary clinical assessment of a large series of females with Rett syndrome (RS; n=87) is presented. Participants' ages ranged from 2 years 1 month to 44 years 10 months. Areas assessed included oromotor skills, feeding problems, growth, breathing abnormalities, mobility, postural abnormalities and joint deformities, epilepsy, hand use and stereotypies, self-care, and cognitive and communication skills. Many previously reported trends in the presentation of RS over time were confirmed, notably the increasingly poor growth and near pervasiveness of fixed joint deformities and scoliosis in adulthood. In contrast, there was a slight trend towards improved autonomic function in adulthood, whereas feeding difficulties increased into middle childhood and then reached a plateau. Improvements in mobility into adolescence were followed by a decline in those skills in adulthood. Levels of dependency were high, confirming findings from previous studies. Despite the presence of repetitive hand movements, a range of hand-use skills was seen in individuals of all ages. Cognitive and communication skills were limited, but there was little evidence of deterioration of these abilities with age. These findings confirm that RS is not a degenerative condition and indicate that intervention and support to maintain and increase motor skills, daily living skills, and cognitive and communicative functioning are appropriate targets for individuals with RS. [ABSTRACT FROM AUTHOR]
- Published
- 2003
- Full Text
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14. Follow-up of children with language delay and features of autism from preschool years to middle childhood.
- Author
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Michelotti J, Charman T, Slonims V, Baird G, Michelotti, Janine, Charman, Tony, Slonims, Vicky, and Baird, Gillian
- Published
- 2002
15. FC14-05 - Parent-mediated communication-focused treatment for preschool children with autism (MRC PACT); a randomised controlled trial
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Green, J., Charman, T., Mc Conachie, H., Aldred, C., Slonims, V., Howlin, P., Le Couteur, A., Leadbitter, K., Hudry, K., Byford, S., Barrett, B., Temple, K., Macdonald, W., and Pickles, A.
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- 2011
- Full Text
- View/download PDF
16. Mediation of 6‐year mid‐childhood follow‐up outcomes after pre‐school social communication (PACT) therapy for autistic children: randomised controlled trial
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Sophie Carruthers, Andrew Pickles, Tony Charman, Helen McConachie, Ann Le Couteur, Vicky Slonims, Patricia Howlin, Rachel Collum, Erica Salomone, Hannah Tobin, Isobel Gammer, Jessica Maxwell, Catherine Aldred, Jeremy Parr, Kathy Leadbitter, Jonathan Green, Carruthers, S, Pickles, A, Charman, T, Mcconachie, H, Le Couteur, A, Slonims, V, Howlin, P, Collum, R, Salomone, E, Tobin, H, Gammer, I, Maxwell, J, Aldred, C, Parr, J, Leadbitter, K, and Green, J
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early intervention ,Psychiatry and Mental health ,Pediatrics, Perinatology and Child Health ,developmental psychopathology ,Developmental and Educational Psychology ,mediation ,Autism spectrum disorder ,structural equation modelling - Abstract
BACKGROUND: There are very few mechanistic studies of the long-term impact of psychosocial interventions in childhood. The parent-mediated Paediatric Autism Communication Therapy (PACT) RCT showed sustained effects on autistic child outcomes from pre-school to mid-childhood. We investigated the mechanism by which the PACT intervention achieved these effects.METHODS: Of 152 children randomised to receive PACT or treatment as usual between 2 and 5 years of age, 121 (79.6%) were followed 5-6 years after the endpoint at a mean age of 10.5 years. Assessors, blind to the intervention group, measured Autism Diagnostic Observation Scale Calibrated Severity Score (ADOS CSS) for child autistic behaviours and Teacher Vineland (TVABS) for adaptive behaviour in school. Hypothesised mediators were child communication initiations with caregivers in a standard play observation (Dyadic Communication Measure for Autism, DCMA). Hypothesised moderators of mediation were baseline child non-verbal age equivalent scores (AE), communication and symbolic development (CSBS) and 'insistence on sameness' (IS). Structural equation modelling was used in a repeated measures mediation design.RESULTS: Good model fits were obtained. The treatment effect on child dyadic initiation with the caregiver was sustained through the follow-up period. Increased child initiation at treatment midpoint mediated the majority (73%) of the treatment effect on follow-up ADOS CSS. A combination of partial mediation from midpoint child initiations and the direct effect of treatment also contributed to a near-significant total effect on follow-up TVABS. No moderation of this mediation was found for AE, CSBS or IS.CONCLUSIONS: Early sustained increase in an autistic child's communication initiation with their caregiver is largely responsible for the long-term effects from PACT therapy on autistic and adaptive behaviour outcomes. This supports the theoretical logic model of PACT therapy but also illuminates fundamental causal processes of social and adaptive development in autism over time: early social engagement in autism can be improved and this can have long-term generalised outcome effects.
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- 2023
17. Randomised trial of a parent-mediated intervention for infants at high risk for autism: longitudinal outcomes to age 3 years
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Green, Jonathan, Pickles, Andrew, Pasco, Greg, Bedford, Rachael, Wan, Ming Wai, Elsabbagh, Mayada, Slonims, Vicky, Gliga, Teea, Jones, Emily, Cheung, Celeste, Charman, Tony, Johnson, Mark, Baron-Cohen, Simon, Bolton, Patrick, Davies, Kim, Liew, Michelle, Fernandes, Janice, Gammer, Isobel, Salomone, Erica, Ribeiro, Helena, Tucker, Leslie, Taylor, Carol, Booth, Rhonda, Harrop, Claire, Holsgrove, Samina, McNally, Janet, Green, J, Pickles, A, Pasco, G, Bedford, R, Wan, M, Elsabbagh, M, Slonims, V, Gliga, T, Jones, E, Cheung, C, Charman, T, Johnson, M, Baron-Cohen, S, Bolton, P, Davies, K, Liew, M, Fernandes, J, Gammer, I, Salomone, E, Ribeiro, H, Tucker, L, Taylor, C, Booth, R, Harrop, C, Holsgrove, S, and Mcnally, J
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Male ,High-risk siblings ,Autism ,Psychological intervention ,high‐risk siblings ,Pediatrics ,Education, Nonprofessional ,Pre‐emptive intervention ,law.invention ,Autism Diagnostic Observation Schedule ,psyc ,0302 clinical medicine ,Randomized controlled trial ,law ,Outcome Assessment, Health Care ,Early Intervention, Educational ,prevention trial ,Developmental and Educational Psychology ,Single-Blind Method ,Longitudinal Studies ,Autism spectrum disorder ,Parent-Child Relations ,parent‐mediated intervention ,high-risk siblings ,Pediatric ,Parenting ,Communication ,Parent-mediated intervention ,05 social sciences ,parent-mediated intervention ,Perinatology and Child Health ,Psychiatry and Mental Health ,Child, Preschool ,Female ,Original Article ,Psychology ,050104 developmental & child psychology ,Clinical psychology ,prevention trials ,Prodromal Symptoms ,autism ,autism spectrum disorder ,Language Development ,Prodrome ,03 medical and health sciences ,high-risk sibling ,Intervention (counseling) ,medicine ,Humans ,Interpersonal Relations ,0501 psychology and cognitive sciences ,Pre-emptive intervention ,Pediatrics, Perinatology and Child Health ,Repeated measures design ,Original Articles ,medicine.disease ,Prevention trials ,030217 neurology & neurosurgery - Abstract
Background: There has been increasing interest in the potential for pre-emptive interventions in the prodrome of autism, but little investigation as to their effect. Methods: A two-site, two-arm assessor-blinded randomised controlled trial (RCT) of a 12-session parent-mediated social communication intervention delivered between 9 and 14 months of age (Intervention in the British Autism Study of Infant Siblings-Video Interaction for Promoting Positive Parenting), against no intervention. Fifty-four infants (28 intervention, 26 nonintervention) at familial risk of autism but not otherwise selected for developmental atypicality were assessed at 9-month baseline, 15-month treatment endpoint, and 27- and 39-month follow-up. Primary outcome: severity of autism prodromal symptoms, blind-rated on Autism Observation Schedule for Infants or Autism Diagnostic Observation Schedule 2nd Edition across the four assessment points. Secondary outcomes: blind-rated parent-child interaction and child language; nonblind parent-rated communication and socialisation. Prespecified intention-to-treat analysis combined estimates from repeated measures within correlated regressions to estimate the overall effect of the infancy intervention over time. Results: Effect estimates in favour of intervention on autism prodromal symptoms, maximal at 27 months, had confidence intervals (CIs) at each separate time point including the null, but showed a significant overall effect over the course of the intervention and follow-up period (effect size [ES] = 0.32; 95% CI 0.04, 0.60; p = .026). Effects on proximal intervention targets of parent nondirectiveness/synchrony (ES = 0.33; CI 0.04, 0.63; p = .013) and child attentiveness/communication initiation (ES = 0.36; 95% CI 0.04, 0.68; p = .015) showed similar results. There was no effect on categorical diagnostic outcome or formal language measures. Conclusions: Follow-up to 3 years of the first RCT of a very early social communication intervention for infants at familial risk of developing autism has shown a treatment effect, extending 24 months after intervention end, to reduce the overall severity of autism prodromal symptoms and enhance parent-child dyadic social communication over this period. We highlight the value of extended follow-up and repeat assessment for early intervention trials.
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- 2017
18. Parent-mediated social communication therapy for young children with autism (PACT): long-term follow-up of a randomised controlled trial
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Pickles, Andrew, Le Couteur, Ann, Leadbitter, Kathy, Salomone, Erica, Cole-Fletcher, Rachel, Tobin, Hannah, Gammer, Isobel, Lowry, Jessica, Vamvakas, George, Byford, Sarah, Aldred, Catherine, Slonims, Vicky, McConachie, Helen, Howlin, Patricia, Parr, Jeremy R, Charman, Tony, Green, Jonathan, Pickles, A, Le Couteur, A, Leadbitter, K, Salomone, E, Cole-Fletcher, R, Tobin, H, Gammer, I, Lowry, J, Vamvakas, G, Byford, S, Aldred, C, Slonims, V, Mcconachie, H, Howlin, P, Parr, J, Charman, T, and Green, J
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Medicine(all) ,Parents ,Behavior Therapy ,Autism Spectrum Disorder ,Medicine (all) ,Communication ,Early Intervention, Educational ,Humans ,Articles ,Autistic Disorder ,Child ,Follow-Up Studies - Abstract
Background It is not known whether early intervention can improve long-term autism symptom outcomes. We aimed to follow-up the Preschool Autism Communication Trial (PACT), to investigate whether the PACT intervention had a long-term effect on autism symptoms and continued effects on parent and child social interaction. Methods PACT was a randomised controlled trial of a parent-mediated social communication intervention for children aged 2–4 years with core autism. Follow-up ascertainment was done at three specialised clinical services centres in the UK (London, Manchester, and Newcastle) at a median of 5·75 years (IQR 5·42–5·92) from the original trial endpoint. The main blinded outcomes were the comparative severity score (CSS) from the Autism Diagnostic Observation Schedule (ADOS), the Dyadic Communication Assessment Measure (DCMA) of the proportion of child initiatiations when interacting with the parent, and an expressive-receptive language composite. All analyses followed the intention-to-treat principle. PACT is registered with the ISRCTN registry, number ISRCTN58133827. Findings 121 (80%) of the 152 trial participants (59 [77%] of 77 assigned to PACT intervention vs 62 [83%] of 75 assigned to treatment as usual) were traced and consented to be assessed between July, 2013, and September, 2014. Mean age at follow-up was 10·5 years (SD 0·8). Group difference in favour of the PACT intervention based on ADOS CSS of log-odds effect size (ES) was 0·64 (95% CI 0·07 to 1·20) at treatment endpoint and ES 0·70 (95% CI −0·05 to 1·47) at follow-up, giving an overall reduction in symptom severity over the course of the whole trial and follow-up period (ES 0·55, 95% CI 0·14 to 0·91, p=0·004). Group difference in DCMA child initiations at follow-up showed a Cohen's d ES of 0·29 (95% CI −0.02 to 0.57) and was significant over the course of the study (ES 0·33, 95% CI 0·11 to 0·57, p=0·004). There were no group differences in the language composite at follow-up (ES 0·15, 95% CI −0·23 to 0·53). Interpretation The results are the first to show long-term symptom reduction after a randomised controlled trial of early intervention in autism spectrum disorder. They support the clinical value of the PACT intervention and have implications for developmental theory. Funding Medical Research Council. Background It is not known whether early intervention can improve long-term autism symptom outcomes. We aimed to follow-up the Preschool Autism Communication Trial (PACT), to investigate whether the PACT intervention had a long-term effect on autism symptoms and continued effects on parent and child social interaction. Methods PACT was a randomised controlled trial of a parent-mediated social communication intervention for children aged 2–4 years with core autism. Follow-up ascertainment was done at three specialised clinical services centres in the UK (London, Manchester, and Newcastle) at a median of 5·75 years (IQR 5·42–5·92) from the original trial endpoint. The main blinded outcomes were the comparative severity score (CSS) from the Autism Diagnostic Observation Schedule (ADOS), the Dyadic Communication Assessment Measure (DCMA) of the proportion of child initiatiations when interacting with the parent, and an expressive-receptive language composite. All analyses followed the intention-to-treat principle. PACT is registered with the ISRCTN registry, number ISRCTN58133827. Findings 121 (80%) of the 152 trial participants (59 [77%] of 77 assigned to PACT intervention vs 62 [83%] of 75 assigned to treatment as usual) were traced and consented to be assessed between July, 2013, and September, 2014. Mean age at follow-up was 10·5 years (SD 0·8). Group difference in favour of the PACT intervention based on ADOS CSS of log-odds effect size (ES) was 0·64 (95% CI 0·07 to 1·20) at treatment endpoint and ES 0·70 (95% CI −0·05 to 1·47) at follow-up, giving an overall reduction in symptom severity over the course of the whole trial and follow-up period (ES 0·55, 95% CI 0·14 to 0·91, p=0·004). Group difference in DCMA child initiations at follow-up showed a Cohen's d ES of 0·29 (95% CI −0.02 to 0.57) and was significant over the course of the study (ES 0·33, 95% CI 0·11 to 0·57, p=0·004). There were no group differences in the language composite at follow-up (ES 0·15, 95% CI −0·23 to 0·53). Interpretation The results are the first to show long-term symptom reduction after a randomised controlled trial of early intervention in autism spectrum disorder. They support the clinical value of the PACT intervention and have implications for developmental theory. Funding Medical Research Council.
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19. Parent-mediated intervention in infants with an elevated likelihood for autism reduces dwell time during a gaze-following task.
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Bedford R, Green J, Gliga T, Jones EH, Elsabbagh M, Pasco G, Wan MW, Slonims V, Charman T, Pickles A, and Johnson MH
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Cognitive markers may in theory be more sensitive to the effects of intervention than overt behavioral measures. The current study tests the impact of the Intervention with the British Autism Study of Infant Siblings-Video Interaction for Promoting Positive Parenting (iBASIS-VIPP) on an eye-tracking measure of social attention: dwell time to the referred object in a gaze following task. The original two-site, two-arm, assessor-blinded randomized controlled trial (RCT) of this intervention to increase parental awareness, and responsiveness to their infant, was run with infants who have an elevated familial likelihood for autism (EL). Fifty-four EL infants (28 iBASIS-VIPP intervention, 26 no intervention) were enrolled, and the intervention took place between 9 months (baseline) and 15 months (endpoint), with gaze following behavior measured at 15 months. Secondary intention to treat (ITT) analysis showed that the intervention was associated with significantly reduced dwell time to the referent of another person's gaze (β = -0.32, SE = 0.14, p = 0.03) at 15-month treatment endpoint. Given the established link between gaze following and language, the results are considered in the context of a previously reported, non-significant and transient trend toward lower language scores at the treatment endpoint (Green et al. (2015) The Lancet Psychiatry, 2(2), 133-140). Future intervention trials should aim to include experimental cognitive measures, alongside behavioral measures, to investigate mechanisms associated with intervention effects., (© 2024 The Author(s). Autism Research published by International Society for Autism Research and Wiley Periodicals LLC.)
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- 2024
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20. Parent-Child Interactions May Help to Explain Relations Between Parent Characteristics and Clinically Observed Child Autistic Behaviours.
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Loncarevic A, Maybery MT, Barbaro J, Dissanayake C, Green J, Hudry K, Iacono T, Slonims V, Varcin KJ, Wan MW, Wray J, and Whitehouse AJO
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- Humans, Male, Female, Cross-Sectional Studies, Infant, Autistic Disorder psychology, Child, Preschool, Adult, Child Behavior psychology, Psychological Distress, Parent-Child Relations, Parents psychology
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The importance of supporting parent-child interactions has been noted in the context of prodromal autism, but little consideration has been given to the possible contributing role of parental characteristics, such as psychological distress. This cross-sectional study tested models in which parent-child interaction variables mediated relations between parent characteristics and child autistic behaviour in a sample of families whose infant demonstrated early signs of autism (N = 103). The findings suggest that associations between parent characteristics (psychological distress; aloofness) and child autistic behaviours may be mediated by the child's inattentiveness or negative affect during interactions. These findings have important implications in developing and implementing interventions in infancy which target the synchrony of parent-child interaction with the goal to support children's social communication development., (© 2023. Crown.)
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- 2024
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21. Generalisation of Social Communication Skills by Autistic Children During Play-Based Assessments Across Home, School and an Unfamiliar Research Setting.
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Carruthers S, Charman T, Leadbitter K, Ellis C, Taylor L, Moore H, Taylor C, James K, Balabanovska M, Langhorne S, Aldred C, Slonims V, Grahame V, Howlin P, McConachie H, Parr J, Emsley R, Le Couteur A, Green J, and Pickles A
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We investigated autistic children's generalisation of social communication over time across three settings during a play-based assessment with different adults and explore the potential moderating effects on generalisation of age, nonverbal IQ and level of restricted and repetitive behaviours. The social communication abilities of 248 autistic children (2-11 years, 21% female, 22% single parent, 60% white) from three UK sites were assessed from 1984 video interactions in three contexts with three different interaction partners (parent/home, teaching assistant/school, researcher/clinic) at baseline, midpoint (+ 7m) and endpoint (+ 12m) within the Paediatric Autism Communication Trial-Generalised (PACT-G), a parent-mediated social communication intervention. Children's midpoint social communication at home generalised to school at midpoint and to clinic at endpoint. Generalisation was stronger from home to school and clinic than school to home and clinic. Generalisation was not moderated by age, nonverbal IQ or restricted and repetitive behaviour. Broader child development did not explain the pattern of results. The current study is the largest study to date to explore generalisation with autistic children and provides novel insight into their generalisation of social communication skills. Further research is needed to gain a more comprehensive understanding of facilitators of generalisation across settings and interaction partners in order to develop targeted strategies for interventions to enhance outcomes for young autistic children., (© 2024. The Author(s).)
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- 2024
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22. 'Turning up and tuning in'. Factors associated with parental non-attendance and non-adherence in intervention for young children with speech, language communication needs.
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Williams P, Slonims V, and Weinman J
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- Child, Female, Humans, Child, Preschool, Communication, Speech Therapy, Mothers, Speech, Parents psychology
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Background: When parents bring their child to appointments and then adhere to agreed speech and language therapy (SLT) recommendations, there is the potential to increase the intensity of the intervention, support generalization and improve outcomes. In SLT, however, little is known about factors that may promote attendance or adherence. Studies in other clinical areas such in medicine, psychology and physiotherapy have identified risk factors for non-attendance or non-adherence that are multifactorial and variable dependent on, for example, population and intervention., Aims: To identify rates of non-attendance and non-adherence, and to identify parent or child factors associated with parent involvement in intervention for children under 5 years of age receiving SLT., Methods: Parents completed questionnaires at two time points assessing the domains of parents' beliefs (problem perceptions, self-efficacy), personal circumstances (socio-demographics, family functioning), treatment experience and child factors. Predictors of parent attendance and adherence were identified through multiple regression analyses. Non-attendance rates were identified via local health records and non-adherence ascertained using a specific parent-reported measure within the treatment experience domain., Results: Participants (N = 199) were predominantly mothers, and were ethnically and socio-economically diverse, speaking a wide range of languages. Their children presented with a range of speech, language communication needs (SLCN). The rate of non-attendance was 25% and the main predictors of non-attendance were maternal age, education level and two factors within the parent beliefs domain. This model explained 40% of the variance in attendance. The rate of non-adherence in this cohort was 26% with parental rating of the importance of a recommendation and self-efficacy beliefs predicting adherence; this explained 56% of the variance in adherence to SLT recommendations at home., Conclusions & Implications: Our research has provided preliminary evidence of the influence of parents' beliefs, personal circumstances and treatment experiences on their involvement in their child's therapy. Speech and language therapists should consider factors impacting attendance and adherence to treatment and explore parental perceptions of their child's SLCN before embarking on an intervention, a foundation for collaborative practice. A possible limitation of this study is that the levels of attrition in our sample led to generally high measured rates of participation, which should be considered in future studies. Future research should explore adherence in treatments with varying doses, with different types of SLCN or interventions and in different settings., What This Paper Adds: What is already known on the subject It is acknowledged that parent involvement in their child's therapy, such as attending and adhering to recommendations, is important but little is known about the rates of involvement and what factors may be associated with attendance and adherence in SLT. Qualitative research has explored parental involvement suggesting that beliefs about an intervention may be pertinent. Extensive research in other clinical areas suggest multiple and varied factors are influential and further exploration of particular populations and interventions is necessary. What this paper adds to the existing knowledge This study identified rates of parental non-attendance and non-adherence in a cohort of predominantly mothers of children under the age of 5 years. It is the first study to measure parent adherence in SLT and identify factors that are associated parental adherence to SLT recommendations. It adds to the small body of SLT specific research in understanding risk factors for non-attendance. What are the potential or actual clinical implications of this work? This study highlights the need for a speech and language therapist to consider and explore parents' perspectives of their child's SLCN as a part of achieving collaboration with a parent in order to achieve the best outcomes. It provides a foundation for further systematic research into parent involvement with the ultimate aim of enhancing outcomes for children with SLCN., (© 2023 The Authors. International Journal of Language & Communication Disorders published by John Wiley & Sons Ltd on behalf of Royal College of Speech and Language Therapists.)
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- 2024
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23. Mediation of 6-year mid-childhood follow-up outcomes after pre-school social communication (PACT) therapy for autistic children: randomised controlled trial.
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Carruthers S, Pickles A, Charman T, McConachie H, Le Couteur A, Slonims V, Howlin P, Collum R, Salomone E, Tobin H, Gammer I, Maxwell J, Aldred C, Parr J, Leadbitter K, and Green J
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- Child, Child, Preschool, Humans, Communication, Follow-Up Studies, Parents, Autism Spectrum Disorder diagnosis, Autistic Disorder therapy, Autistic Disorder psychology
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Background: There are very few mechanistic studies of the long-term impact of psychosocial interventions in childhood. The parent-mediated Paediatric Autism Communication Therapy (PACT) RCT showed sustained effects on autistic child outcomes from pre-school to mid-childhood. We investigated the mechanism by which the PACT intervention achieved these effects., Methods: Of 152 children randomised to receive PACT or treatment as usual between 2 and 5 years of age, 121 (79.6%) were followed 5-6 years after the endpoint at a mean age of 10.5 years. Assessors, blind to the intervention group, measured Autism Diagnostic Observation Scale Calibrated Severity Score (ADOS CSS) for child autistic behaviours and Teacher Vineland (TVABS) for adaptive behaviour in school. Hypothesised mediators were child communication initiations with caregivers in a standard play observation (Dyadic Communication Measure for Autism, DCMA). Hypothesised moderators of mediation were baseline child non-verbal age equivalent scores (AE), communication and symbolic development (CSBS) and 'insistence on sameness' (IS). Structural equation modelling was used in a repeated measures mediation design., Results: Good model fits were obtained. The treatment effect on child dyadic initiation with the caregiver was sustained through the follow-up period. Increased child initiation at treatment midpoint mediated the majority (73%) of the treatment effect on follow-up ADOS CSS. A combination of partial mediation from midpoint child initiations and the direct effect of treatment also contributed to a near-significant total effect on follow-up TVABS. No moderation of this mediation was found for AE, CSBS or IS., Conclusions: Early sustained increase in an autistic child's communication initiation with their caregiver is largely responsible for the long-term effects from PACT therapy on autistic and adaptive behaviour outcomes. This supports the theoretical logic model of PACT therapy but also illuminates fundamental causal processes of social and adaptive development in autism over time: early social engagement in autism can be improved and this can have long-term generalised outcome effects., (© 2023 The Authors. Journal of Child Psychology and Psychiatry published by John Wiley & Sons Ltd on behalf of Association for Child and Adolescent Mental Health.)
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- 2024
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24. Clinical and cost-effectiveness of an adapted intervention for preschoolers with moderate to severe intellectual disabilities displaying behaviours that challenge: the EPICC-ID RCT.
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Ondruskova T, Royston R, Absoud M, Ambler G, Qu C, Barnes J, Hunter R, Panca M, Kyriakopoulos M, Oulton K, Paliokosta E, Sharma AN, Slonims V, Summerson U, Sutcliffe A, Thomas M, Dhandapani B, Leonard H, and Hassiotis A
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- Child, Preschool, Humans, Cost-Benefit Analysis, London, Quality of Life, Single-Blind Method, COVID-19, Intellectual Disability
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Background: Stepping Stones Triple P is an adapted intervention for parents of young children with developmental disabilities who display behaviours that challenge, aiming at teaching positive parenting techniques and promoting a positive parent-child relationship., Objective: To evaluate the clinical and cost-effectiveness of level 4 Stepping Stones Triple P in reducing behaviours that challenge in children with moderate to severe intellectual disabilities., Design, Setting, Participants: A parallel two-arm pragmatic multisite single-blind randomised controlled trial recruited a total of 261 dyads (parent and child). The children were aged 30-59 months and had moderate to severe intellectual disabilities. Participants were randomised, using a 3 : 2 allocation ratio, into the intervention arm (Stepping Stones Triple P; n = 155) or treatment as usual arm ( n = 106). Participants were recruited from four study sites in Blackpool, North and South London and Newcastle., Intervention: Level 4 Stepping Stones Triple P consists of six group sessions and three individual phone or face-to-face contacts over 9 weeks. These were changed to remote sessions after 16 March 2020 due to the coronavirus disease 2019 pandemic., Main Outcome Measure: The primary outcome measure was the parent-reported Child Behaviour Checklist, which assesses the severity of behaviours that challenge., Results: We found a small non-significant difference in the mean Child Behaviour Checklist scores (-4.23, 95% CI -9.98 to 1.52, p = 0.146) in the intervention arm compared to treatment as usual at 12 months. Per protocol and complier average causal effect sensitivity analyses, which took into consideration the number of sessions attended, showed the Child Behaviour Checklist mean score difference at 12 months was lower in the intervention arm by -10.77 (95% CI -19.12 to -2.42, p = 0.014) and -11.53 (95% CI -26.97 to 3.91, p = 0.143), respectively. The Child Behaviour Checklist mean score difference between participants who were recruited before and after the coronavirus disease 2019 pandemic was estimated as -7.12 (95% CI -13.44 to -0.81) and 7.61 (95% CI -5.43 to 20.64), respectively ( p = 0.046), suggesting that any effect pre-pandemic may have reversed during the pandemic. There were no differences in all secondary measures. Stepping Stones Triple P is probably value for money to deliver (-£1057.88; 95% CI -£3218.6 to -£46.67), but decisions to roll this out as an alternative to existing parenting interventions or treatment as usual may be dependent on policymaker willingness to invest in early interventions to reduce behaviours that challenge. Parents reported the intervention boosted their confidence and skills, and the group format enabled them to learn from others and benefit from peer support. There were 20 serious adverse events reported during the study, but none were associated with the intervention., Limitations: There were low attendance rates in the Stepping Stones Triple P arm, as well as the coronavirus disease 2019-related challenges with recruitment and delivery of the intervention., Conclusions: Level 4 Stepping Stones Triple P did not reduce early onset behaviours that challenge in very young children with moderate to severe intellectual disabilities. However, there was an effect on child behaviours for those who received a sufficient dose of the intervention. There is a high probability of Stepping Stones Triple P being at least cost neutral and therefore worth considering as an early therapeutic option given the long-term consequences of behaviours that challenge on people and their social networks., Future Work: Further research should investigate the implementation of parenting groups for behaviours that challenge in this population, as well as the optimal mode of delivery to maximise engagement and subsequent outcomes., Study Registration: This study is registered as NCT03086876 (https://www.clinicaltrials.gov/ct2/show/NCT03086876?term=Hassiotis±Angela&draw=1&rank=1)., Funding: This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: HTA 15/162/02) and is published in full in Health Technology Assessment ; Vol. 28, No. 6. See the NIHR Funding and Awards website for further award information.
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- 2024
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25. Factors associated with mental health symptoms among UK autistic children and young people and their parents during the COVID-19 pandemic.
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Palmer M, Chandler S, Carter Leno V, Mgaieth F, Yorke I, Hollocks M, Pickles A, Slonims V, Scott S, Charman T, and Simonoff E
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- Adolescent, Humans, Child, Mental Health, Pandemics, Parents psychology, United Kingdom epidemiology, COVID-19, Autistic Disorder diagnosis, Autism Spectrum Disorder epidemiology, Autism Spectrum Disorder complications
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Lay Abstract: What is already known about the topic: The COVID-19 pandemic and the associated restrictions impacted all of society. There is emerging evidence showing a range of impacts on autistic children and young people and their families. Further research that looks at how individuals coped during the pandemic while considering how they were doing before the pandemic is needed. What this paper adds: This article explores whether how well autistic youth were doing before the pandemic influenced how they coped during the pandemic. It also looked at how well their parents were doing during the pandemic and whether any pre-pandemic factors influenced how they coped. Samples of both primary-school-aged autistic children and autistic teenagers and their parents were surveyed to answer these questions. More engagement and enjoyment in education provision during the pandemic and getting outside more were linked with better child and parental mental health during the pandemic. More attention deficit hyperactivity disorder before the pandemic was linked with more attention deficit hyperactivity disorder and behavioural problems during the pandemic in primary-school-aged autistic children, and more emotional problems during the pandemic in autistic teenagers. Parents with more mental health problems during the pandemic had more mental health problems before the pandemic. Implications for practice, research or policy: Encouraging engagement and enjoyment in education and promoting physical exercise are key intervention targets. Ensuring access to attention deficit hyperactivity disorder medication and support is important, especially if this is managed jointly across school and home.
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- 2023
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26. Effects of a Parenting Intervention for Emotional and Behavioral Problems in Young Autistic Children Under Conditions of Enhanced Uncertainty: Two-Year Follow-up of a Pilot Randomized Controlled Trial Cohort (ASTAR) During the United Kingdom COVID-19 Pandemic.
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Palmer M, Carter Leno V, Hallett V, Mueller JM, Breese L, Pickles A, Slonims V, Scott S, Charman T, and Simonoff E
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- Humans, Child, Parenting, Follow-Up Studies, Uncertainty, Pilot Projects, Communicable Disease Control, Parents psychology, United Kingdom, Autistic Disorder, Problem Behavior, COVID-19
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Objective: Most young autistic children display emotional and behavioral problems (EBPs). There is evidence that behavioral parenting interventions (BPIs) reduce these. The COVID-19 pandemic and associated lockdowns can be seen as a natural experiment to test the longer-term effect of BPIs under conditions of increased uncertainty., Method: Opportunistic follow-up (n = 49) of a pilot randomized controlled trial (RCT) cohort (n = 62 autistic children aged 6-11 years; originally randomized to a 12-week group BPI [Predictive Parenting; n = 31] or an attention control [Psychoeducation; n = 31]) was conducted during COVID-19-related lockdowns. Measures of parent-reported child irritability and parenting stress were collected at 3 time points (baseline: mean age = 6.7 years; primary endpoint: mean age = 7.1 years, ∼5 months after randomization; and COVID-19 follow-up: mean age = 8.8 years, ∼2 years after randomization). We tested the magnitude of intervention effects using point estimates of differences in child irritability and parenting stress between arms at primary endpoint and COVID-19 follow-up, covarying for baseline scores. We used area under the curve (AUC) analyses to obtain overall estimates of the average intervention effect across all 3 timepoints. Semi-structured qualitative interviews were conducted with a subsample of parents (n = 18)., Results: A small but significant intervention effect was found from baseline to COVID-19 follow-up in favor of Predictive Parenting on parent-reported child irritability (d = -0.33, 95% CI = -0.65, -0.01) and parenting stress (d = -0.31, 95% CI = -0.59, -0.03). No overall mean intervention effect for these measures as estimated by the AUC analyses (which takes into account the nonsignificant effect at primary endpoint) was found. Interview feedback on the both interventions was positive, and parents reported using strategies from Predictive Parenting during COVID-19-related restrictions., Conclusion: This opportunistic follow-up study at a time of stress indicates the need for careful consideration of how and when to measure the effects of BPIs in autistic child populations. Future trials should consider both the most appropriate endpoint and in what context effects may be more likely to be seen., Clinical Trial Registration Information: Autism Spectrum Treatment and Resilience (ASTAR); https://www.isrctn.com; 91411078., (Crown Copyright © 2022. Published by Elsevier Inc. All rights reserved.)
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- 2023
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27. Using implementation science frameworks to explore barriers and facilitators for parents' use of therapeutic strategies following a parent-mediated autism intervention.
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Carruthers S, Mleczko N, Page S, Ahuja S, Ellis C, Howlin P, Leadbitter K, Taylor L, Slonims V, and Charman T
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- Child, Humans, Implementation Science, Parents, Communication, Autistic Disorder therapy, Autism Spectrum Disorder therapy
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Lay Abstract: Many early autism interventions teach parents therapeutic strategies to help them adjust their communication style with their children. Research has shown that this behaviour change in parents leads to improvements in child communication. It is, therefore, important to learn what factors support or hinder parents in their use of therapeutic strategies learned in such interventions. This study set out to interview parents who had participated in a research trial of the Paediatric Autism Communication Therapy-Generalised intervention. We interviewed 27 caregivers and explored their use of the strategies up to 2 years after the end of the research trial. Qualitative frameworks were used to inform interview questions and data analysis. These frameworks focused on a range of contextual factors, including parents' characteristics, their context and features of the intervention. Parents reported barriers and facilitators to using Paediatric Autism Communication Therapy-Generalised strategies across three themes: Motivating Factors; Opportunity and Support; Parent Characteristics. One of these themes, Motivating Factors, was further divided into the subthemes Compatibility and Buy-In and Alignment of Goals and Outcomes. Almost all parents reported continued use of the Paediatric Autism Communication Therapy-Generalised strategies. Facilitators included parental confidence in using the strategies and barriers included child's behaviour. Consideration of these factors can inform ways to better support parents in future autism interventions.
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- 2023
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28. The Utility of Natural Language Samples for Assessing Communication and Language in Infants Referred with Early Signs of Autism.
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Hudry K, Smith J, Pillar S, Varcin KJ, Bent CA, Boutrus M, Chetcuti L, Clark A, Dissanayake C, Iacono T, Kennedy L, Lant A, Robinson Lake J, Segal L, Slonims V, Taylor C, Wan MW, Green J, and Whitehouse AJO
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- Child, Humans, Infant, Reproducibility of Results, Communication, Language, Child Language, Autistic Disorder diagnosis
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Natural Language Sampling (NLS) offers clear potential for communication and language assessment, where other data might be difficult to interpret. We leveraged existing primary data for 18-month-olds showing early signs of autism, to examine the reliability and concurrent construct validity of NLS-derived measures coded from video-of child language, parent linguistic input, and dyadic balance of communicative interaction-against standardised assessment scores. Using Systematic Analysis of Language Transcripts (SALT) software and coding conventions, masked coders achieved good-to-excellent inter-rater agreement across all measures. Associations across concurrent measures of analogous constructs suggested strong validity of NLS applied to 6-min video clips. NLS offers benefits of feasibility and adaptability for validly quantifying emerging skills, and potential for standardisation for clinical use and rigorous research design., (© 2023. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.)
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29. Caregiver sensitivity predicts infant language use, and infant language complexity predicts caregiver language complexity, in the context of possible emerging autism.
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Smith J, Chetcuti L, Kennedy L, Varcin KJ, Slonims V, Bent CA, Green J, Iacono T, Pillar S, Taylor C, Wan MW, Whitehouse AJO, and Hudry K
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- Child, Humans, Infant, Caregivers, Prospective Studies, Language, Language Development, Autistic Disorder, Autism Spectrum Disorder
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While theory supports bidirectional effects between caregiver sensitivity and language use, and infant language acquisition-both caregiver-to-infant and also infant-to-caregiver effects-empirical research has chiefly explored the former unidirectional path. In the context of infants showing early signs of autism, we investigated prospective bidirectional associations with 6-min free-play interaction samples collected for 103 caregivers and their infants (mean age 12-months; and followed up 6-months later). We anticipated that measures of caregiver sensitivity/language input and infant language would show within-domain temporal stability/continuity, but also that there would be predictive associations from earlier caregiver input to subsequent child language, and vice versa. Caregiver sensitive responsiveness (from the Manchester Assessment of Caregiver-Infant interaction [MACI]) predicted subsequent infant word tokens (i.e., amount of language, coded following the Systematic Analysis of Language Transcripts [SALT]). Further, earlier infant Mean Length of Utterance (MLU; reflecting language complexity, also derived from SALT coding) predicted later caregiver MLU, even when controlling for variability in infant ages and clear within-domain temporal stability/continuity in key measures (i.e., caregiver sensitive responsiveness and infant word tokens; and infant and caregiver MLU). These data add empirical support to theorization on how caregiver input can be both supportive of, and potentially influenced by, infant capacities, when infants have social-communication differences and/or communication/language delays suggestive of possible emerging autism., (© 2022 The Authors. Autism Research published by International Society for Autism Research and Wiley Periodicals LLC.)
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- 2023
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30. Parent, Teacher and Observational Reports of Emotional and Behavioral Problems in Young Autistic Children.
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Palmer M, Tarver J, Carter Leno V, Paris Perez J, Frayne M, Slonims V, Pickles A, Scott S, Charman T, and Simonoff E
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- Humans, Parents psychology, Parenting psychology, Problem Behavior, Autistic Disorder, Autism Spectrum Disorder diagnosis
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Emotional and behavioral problems (EBPs) frequently occur in young autistic children. Discrepancies between parents and other informants are common but can lead to uncertainty in formulation, diagnosis and care planning. This study aimed to explore child and informant characteristics are associated with reported child EBPs across settings. Participants were 83 4-8-year-old autistic children and their parents and teachers in the Autism Spectrum Treatment and Resilience (ASTAR) study. Questionnaires of child EBPs were completed by parents and teachers, and self-reported parenting stress and wellbeing measures were obtained. An observation of parent-child/researcher-child interaction was also completed. Parents reported more EBPs than teachers and parent-teacher agreement was low, particularly for emotional problems. Greater parenting stress and being verbal was associated with more parent- but not teacher-reported EBPs. More observed behaviors that challenge were displayed by minimally verbal children. More parenting stress could be associated with the presence of more EBPs in the home; alternatively, parenting stress may confound reports. It is essential for assessments of EBPs in autistic children to take a multi-informant approach. Better understanding of the associations between informant characteristics and informant discrepancies of EBPs is needed., (© 2022. The Author(s).)
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- 2023
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31. An adapted social communication intervention at home and education to promote social communication change in children with severe autism: the PACT-G RCT
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Green J, Leadbitter K, Ellis C, Taylor L, Moore HL, Carruthers S, James K, Taylor C, Balabanovska M, Langhorne S, Aldred C, Slonims V, Grahame V, Parr J, Humphrey N, Howlin P, McConachie H, Le Couteur A, Charman T, Emsley R, and Pickles A
- Abstract
Background: Prior evidence suggests that behaviours closely related to the intervention delivered for autism are amenable to change, but it becomes more difficult when generalising treatment effects beyond that immediate context., Objectives: The objectives were (1) to test an early autism social communication intervention designed to promote child social communication change in the naturalistic contexts of both home and education, with an additive effect on overall child symptom outcomes, and (2) to conduct a mechanistic study investigating the transmission of treatment effects within and across contexts to an overall treatment effect., Design: The trial was a three-site, parallel-group, randomised controlled trial of the experimental treatment plus treatment as usual and treatment as usual alone. The primary intention-to-treat analysis used analysis of covariance. The mechanism analysis used regression models to test mediation of the primary outcome by parent–child and education staff (learning support assistant)–child social interactions., Setting: The study took place in three urban/semiurban regions in Manchester, Newcastle upon Tyne and London., Participants: Children aged 2–11 years who met the criteria for severe autism., Interventions: The Preschool Autism Communication Trial was adapted to parallel components within home and educational settings using in-person and remote delivery. Treatment as usual was the control condition., Main Outcome Measures: The primary outcome was autism symptoms on the Autism Diagnostic Observation schedule-2. The secondary outcomes were Brief Observation of Social Communication Change, dyadic social interaction between child and parent or learning support assistant, reported language, functional outcome and reduction in child disruptive behaviour. Outcomes were measured at baseline and at the 12-month end point in all settings; interim mechanism measurements were taken at 7 months., Results: Participants ( n = 249; 122 in the PACT-G group and 127 in the treatment-as-usual group; 51 were female and 197 were male) received a median of 10 (interquartile range 8–12) sessions at home and 8 (interquartile range 5–10) sessions in an educational setting. We found no significant treatment effects on the end-point Autism Diagnostic Observation Schedule-2 primary outcome (–0.04, 95% confidence interval –0.26 to 0.18; p = 0.734), on the end-point Brief Observation of Social Communication Change secondary outcome (–0.03, 95% confidence interval –0.31 to 0.25; p = 0.85) or on language, repetitive behaviour, adaptive behaviour and child well-being. We did find significant treatment effects on dyadic interactions (increased parent synchronous response 0.54, 95% confidence interval 0.39 to 0.69; p = 0.001); child initiations with a parent (0.27, 95% confidence interval 0.12 to 0.41; p = 0.001); learning support assistant synchronous response (0.32, 95% confidence interval 0.14 to 0.49; p = 0.001); child initiations with a learning support assistant (0.21, 95% confidence interval 0.06 to 0.36; p = 0.005); and unblinded measures of improved parental well-being and child disruptive behaviour across home and educational settings. Adult (parent/learning support assistant) synchronous responsiveness in a home/education setting improved child dyadic social initiation. The child dyadic social initiation was also associated with child symptoms on researcher Brief Observation of Social Communication Change., Limitations: The delivered sessional dosage was 83% of that planned in the home setting and 67% in the educational setting, with 5.5% of home sessions and 5% of educational sessions deemed ‘unacceptable’, particularly for remote delivery. A change of therapy learning support assistant was experienced by over one-third of children by the mid-point of the trial, by another third by the end point, and by one-fifth at both points., Conclusions: The multicomponent Paediatric Autism Communication Trial – Generalised (PACT-G) treatment for a child in a home or educational setting did not produce the hypothesised improvement in child autism symptomatology or adaptive behaviour, but did produce significant improvements in proximal adult–child reciprocal dyadic communication., Future Work: Future work will involve building on these results towards a further understanding of delivery options, dosage and multicomponent extension of social communication interventions for young children with autism in naturalistic settings., Trial Registration: Current Controlled Trials ISRCTN25378536., Funding: This project was funded by the Efficacy and Mechanism Evaluation programme, a Medical Research Council and National Institute for Health and Care Research (NIHR) partnership. This was also part funded by the NIHR Biomedical Research Centre at South London and Maudsley NHS Foundation Trust and King’s College London. This will be published in full in Efficacy and Mechanism Evaluation ; Vol. 9, No. 3. See the NIHR Journals Library website for further project information., (Copyright © 2022 Green et al. This work was produced by Green et al. under the terms of a commissioning contract issued by the Secretary of State for Health and Social Care. This is an Open Access publication distributed under the terms of the Creative Commons Attribution CC BY 4.0 licence, which permits unrestricted use, distribution, reproduction and adaption in any medium and for any purpose provided that it is properly attributed. See: https://creativecommons.org/licenses/by/4.0/. For attribution the title, original author(s), the publication source – NIHR Journals Library, and the DOI of the publication must be cited.)
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32. Combined social communication therapy at home and in education for young autistic children in England (PACT-G): a parallel, single-blind, randomised controlled trial.
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Green J, Leadbitter K, Ellis C, Taylor L, Moore HL, Carruthers S, James K, Taylor C, Balabanovska M, Langhorne S, Aldred C, Slonims V, Grahame V, Parr J, Humphrey N, Howlin P, McConachie H, Couteur AL, Charman T, Emsley R, and Pickles A
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- Adult, Child, Child, Preschool, Female, Humans, Male, Communication, England, Quality of Life, Single-Blind Method, Treatment Outcome, Autistic Disorder psychology, Autistic Disorder therapy
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Background: Autistic children can have difficulty generalising treatment effects beyond the immediate treatment context. Paediatric Autism Communication Therapy (PACT) has been successful when delivered in the clinic. Here we tested the Paediatric Autism Communication Therapy-Generalised (PACT-G) intervention combined between home and education settings for its overall effect and mechanistic transmission of effect across contexts., Methods: In this parallel, single-blind, randomised, controlled trial, we recruited autistic children aged 2-11 years in urban or semi-urban areas in Manchester, Newcastle, and London, England. Children needed to meet core autism criteria on Autism Diagnostic Observation Schedule-second edition (ADOS-2) and parent-rated Social Communication Questionnaire (SCQ-lifetime), and children older than 5 years were included if they had intentional communication but expressive language equivalent of age 4 years or younger. Eligible children were randomly assigned (1:1), using block randomisation (random block sizes of 2 and 4) and stratified for site, age (2-4 years vs 5-11 years), and gender, to either PACT-G plus treatment as usual or treatment as usual alone. Research assessors were masked to treatment allocation. The PACT-G intervention was delivered by a therapist in parallel to the child's parents at home and to learning-support assistants (LSA) at their place of education, using both in-person and remote sessions over a 6 month period, to optimise adult-child social interaction. Treatment as usual included any health support or intervention from education or local community services. The primary outcome was autism symptom severity using the ADOS-2, as measured by researchers, at 12 months versus baseline. Secondary outcomes were Brief Observation of Social Communication Change (BOSCC) and dyadic social interaction between child and adult across contexts, both at 12 months. Other secondary outcome measures were assessed using the following composites: language, anxiety, repetitive behaviour, adaptive behaviour, parental wellbeing, child health-related quality of life, and disruptive behaviour. Assessments were done at baseline, 7 months, and 12 months. We used an intention-to-treat (ITT) analysis of covariance for the efficacy outcome measures. Adverse events were assessed by researchers for all trial families at each contact and by therapists in the PACT-G group at each visit. This study is registered with the ISRCTN Registry, ISRCTN 25378536., Findings: Between Jan 18, 2017, and April 19, 2018, 555 children were referred and 249 were eligible, agreed to participate, and were randomly assigned to either PACT-G (n=122) or treatment as usual (n=127). One child in the PACT-G group withdrew and requested their data be removed from the study, giving an ITT population of 248 children. 51 (21%) of 248 children were female, 197 (79%) were male, 149 (60%) were White, and the mean age was 4·0 years (SD 0·6). The groups were well balanced for demographic and clinical characteristics. In the PACT-G group, parents of children received a median of 10 (IQR 8-12) home sessions and LSAs received a median of 8 (IQR 5-10) education sessions over 6 months. We found no treatment effect on the ADOS-2 primary outcome compared with treatment as usual (effect size 0·04 [95% CI -0·19 to 0·26]; p=0·74), or researcher-assessed BOSCC (0·03 [-0·25 to 0·31]), language composite (-0·03 [-0·15 to 0·10]), repetitive behaviour composite (0·00 [-0·35 to 0·35]), adaptive behaviour composite (0·01 [-0·15 to 0·18]), or child wellbeing (0·09 [-0·15 to 0·34]). PACT-G treatment improved synchronous response in both parent (0·50 [0·36 to 0·65]) and LSA (0·33 [0·16 to 0·50]), mediating increased child communication with parent (0·26 [0·12 to 0·40]) and LSA (0·20 [0·06 to 0·34]). Child dyadic communication change mediated outcome symptom alteration on BOSCC at home (indirect effect -0·78 [SE 0·34; 95% CI -1·44 to -0·11]; p=0·022) although not in education (indirect effect -0·67 [SE 0·37; 95% CI -1·40 to 0·06]; p=0·073); such an effect was not seen on ADOS-2. Treatment with PACT-G also improved the parental wellbeing composite (0·44 [0·08 to 0·79]) and the child disruptive behaviour composite in home and education (0·29 [0·01 to 0·57]). Adverse events on child behaviour and wellbeing were recorded in 13 (10%) of 127 children in the treatment as usual group (of whom four [31%] were girls) and 11 (9%) of 122 in the PACT-G group (of whom three [33%] were girls). One serious adverse event on parental mental health was recorded in the PACT-G group and was possibly study related., Interpretation: Although we found no effect on the primary outcome compared with treatment as usual, adaptation of the 12-month PACT intervention into briefer multicomponent delivery across home and education preserved the positive proximal outcomes, although smaller in effect size, and the original pattern of treatment mediation seen in clinic-delivered therapy, as well as improving parental wellbeing and child disruptive behaviours across home and school. Reasons for this reduced efficacy might be the reduced dose of each component, the effect of remote delivery, and the challenges of the delivery contexts. Caution is needed in assuming that changing delivery methods and context will preserve an original intervention efficacy for autistic children., Funding: National Institute for Health Research and Medical Research Council Efficacy and Mechanism Evaluation Award., Competing Interests: Declaration of interests JG and AP are National Institute for Health Research (NIHR) senior investigators (NIHR NF-SI-0617-10168 for JG and NF-SI-0617-10120 for AP). RE is supported by an NIHR Research Professorship (NIHR300051) and is a member of NIHR Clinical Trials Unit Standing Advisory Committee and Health Technology Assessment Clinical Evaluation and Trials Committee. JG and CA receive director's fees from a not-for-profit PACT training company IMPACT (CiC 10902031). SC is funded by the UK Medical Research Council (MRC). TC has served as a paid consultant to F Hoffmann-La Roche and Servier, and has received royalties from Sage Publications and Guilford Publications. AP receives book and questionnaire royalties from Western Psychological Services, Oxford University Press, and Imperial College Press. All other authors declare no competing interests., (Copyright © 2022 The Author(s). Published by Elsevier Ltd. This is an Open Access article under the CC BY 4.0 license. Published by Elsevier Ltd.. All rights reserved.)
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33. The Lancet Commission on the future of care and clinical research in autism.
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Lord C, Charman T, Havdahl A, Carbone P, Anagnostou E, Boyd B, Carr T, de Vries PJ, Dissanayake C, Divan G, Freitag CM, Gotelli MM, Kasari C, Knapp M, Mundy P, Plank A, Scahill L, Servili C, Shattuck P, Simonoff E, Singer AT, Slonims V, Wang PP, Ysrraelit MC, Jellett R, Pickles A, Cusack J, Howlin P, Szatmari P, Holbrook A, Toolan C, and McCauley JB
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- Autistic Disorder epidemiology, Global Burden of Disease, Humans, Autistic Disorder therapy, Biomedical Research trends, Global Health, Health Services Needs and Demand
- Abstract
Competing Interests: Declaration of interests CL reports royalties from Western Psychological Services for diagnostic instruments; is on scientific advisory boards or projects for Tilray, Roche, Gateway, Springtide, and Greenwich Biosciences; and is supported by grants from the US National Institute of Mental Health (R01HD081199, R01MH115363, R01MH104423–02, and R01MH114925). TCh reports consultancy fees from Roche and Servier; royalties from Sage Publications and Guilford Publications; grants from the UK Medical Research Council (grants number MR/K021389/1 and MR/R011427/1), the UK National Institute for Health Research (grants number 13/119/18 and RP-PG-1211–20016), the EU Horizon 2020 programme (grant number 814302), Innovative Medicines Initiative (grant agreement number 777394 and AIMS-2-TRIALS, which receives support from the EU Horizon 2020 programme, European Federation of Pharmaceutical Industries and Associations, Autism Speaks, Autistica, and Simons Foundation Autism Research Initiative); and grant support from MQ Mental Health Research, The Waterloo Foundation, Epilepsy Research UK, the Charles Hawkins Fund, and the Baily Thomas Foundation. EA reports consulting fees from Roche and Quadrant; funding from Roche and SynapDx; non-financial support from AMO Pharma; book royalties from APPI and Springer; editorial board honoraria from Wiley; a patent (US20160000365A1) for an anxiety meter; is supported by the Ontario Brain Institute (POND network), Canadian Institutes of Health Research, Autism Intervention Research Network on Physical Health (funded by the Health Resources and Services Administration), US National Institutes of Health, Brain Canada, and the Azrieli Foundation; and is the Canada Research Chair in translational therapeutics in autism spectrum disorder and Dr Stuart D Sims endowed chair in Autism. BB is supported by US National Institute of Mental Health grant (1R01HD82127). AHa was supported by grants from the South-Eastern Norway Regional Health Authority (grant numbers 2018059 and 2020022) and by the Research Council of Norway (grants number 274611 and 288083). RJ reports funding from Australia's National Health and Medical Research Council. JC is supported by National Institute of Mental Health grants NIMH09902, NIMH081148, NIMH083707, NIMH80011, and U10MH66764. PC is supported by Simons Foundation Powering Autism Research and the Maternal and Child Health Bureau of the Leadership Education in Neurodevelopmental Disability. TCa reports personal fees from Western Psychological Services. LS reports being a consultant for Roche, Yamo, Teva, Impel, and Janssen Pharmaceuticals; book royalties from Oxford University Press, Guilford Press, and American Psychological Association Press; and licensing royalties for the Parent-Rated Anxiety Scale for Autism Spectrum Disorder from Cogstate and Yamo. PPW reports being employed by Clinical Research Associates, a non-profit organisation that is developing an experimental drug treatment for autism. CMF receives royalties for books on autism spectrum disorder, attention-deficit hyperactivity disorder, and major depressive disorder; grants for the A-FIPP randomised controlled trial (FR2069/8–1, 8-2), awarded by the German Research Foundation, and for the EU-AIMS2-TRIALS (H2020 777394) and EU STIPED (H2020 731827), awarded by the EU. CK is supported by grants from the Health Resources and Services Administration of the US Department of Health and Human Services (UA3MC11055), the US National Institutes of Health (R01HD098248, R01HD095973, P50DC018006), the US Department of Defense, and the Goldman Foundation (3104). APi receives questionnaire royalties from WPS. AHo reports personal fees from GW Pharmaceuticals. PSz receives research funding from the Canadian Institutes of Health Research and royalties from Guilford Press and Simon & Schuster for books. PSh reports support from the Health Resources and Services Administration of the US Department of Health and Human Services under the Autism Transitions Research Project (UJ2MC31073). VS is supported by the National Institute for Health Research (grant numbers EME 13/119/18, RP-PG-1211–20016, and 15/162) and a grant from Autistica (number 7262). GD reports grants from the UK Medical Research Council and Grand Challenges Canada, outside the submitted work. All other authors declare no competing interests.
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34. Caregiver Psychological Distress Predicts Temperament and Social-Emotional Outcomes in Infants with Autism Traits.
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Chetcuti L, Uljarević M, Varcin KJ, Boutrus M, Pillar S, Dimov S, Barbaro J, Dissanayake C, Green J, Wan MW, Segal L, Slonims V, Whitehouse AJO, and Hudry K
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- Aged, Caregivers, Child, Emotions, Humans, Infant, Temperament, Autistic Disorder, Psychological Distress
- Abstract
Child temperament and caregiver psychological distress have been independently associated with social-emotional difficulties among individuals with autism. However, the interrelationship among these risk factors has rarely been investigated. We explored the reciprocal interplay between child temperament (surgency, negative affectivity, and self-regulation) and caregiver psychological distress in the development of child internalizing and externalizing symptoms, in a cohort of 103 infants showing early autism traits. Caregivers completed questionnaires when children were aged around 12-months (Time 1 [T1]), 18-months (Time 2 [T2]), and 24-months (Time 3 [T3]). Cross-lagged path models revealed a significant pathway from T1 caregiver psychological distress through lower T2 child self-regulation to subsequently greater T3 child internalizing symptoms. No such caregiver-driven pathway was evident through T2 child negative affectivity or in the prediction of T3 child externalizing symptoms. Further, no support was found for temperament-driven pathways through caregiver psychological distress to child social-emotional difficulties. Child surgency was mostly unrelated to caregiver psychological distress and social-emotional difficulties. These findings implicate the need to support the mental health of caregivers with an infant with autism traits in order to enhance the emotion regulation and social-emotional development of their infants., (© 2021. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.)
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- 2021
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35. A Novel Group Parenting Intervention for Emotional and Behavioral Difficulties in Young Autistic Children: Autism Spectrum Treatment and Resilience (ASTAR): A Randomized Controlled Trial.
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Charman T, Palmer M, Stringer D, Hallett V, Mueller J, Romeo R, Tarver J, Paris Perez J, Breese L, Hollett M, Cawthorne T, Boadu J, Salazar F, O'Leary M, Beresford B, Knapp M, Slonims V, Pickles A, Scott S, and Simonoff E
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- Child, Emotions, Humans, Parenting, Parents, Autistic Disorder, Problem Behavior
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Objective: To examine the feasibility and preliminary efficacy of a group behavioral parenting intervention for emotional and behavioral problems (EBPs) in young autistic children., Method: This was a feasibility pilot randomized controlled trial comparing a 12-week group behavioral parenting intervention (Predictive Parenting) to an attention control (Psychoeducation). Parents of 62 autistic children 4 to 8 years of age were randomized to Predictive Parenting (n = 31) or Psychoeducation (n = 31). The primary outcome was a blinded observational measure of child behaviors that challenge. Secondary outcomes were observed child compliance and parenting behaviors; parent- and teacher-reported child EBPs; self-reported parenting practices, stress, self-efficacy, and well-being. Cost-effectiveness was also explored., Results: Recruitment, retention, completion of measures, treatment fidelity, and parental satisfaction were high for both interventions. There was no group difference in primary outcome: mean log of rate 0.18 lower (d, 90% CI = -0.44 to 0.08) in Predictive Parenting. Differences in rates of child compliance (0.44, 90% CI = 0.11 to 0.77), facilitative parenting (0.63, 90% CI = 0.33 to 0.92) and parent-defined target symptom change (-0.59, 90% CI -0.17 to -1.00) favored Predictive Parenting. There were no differences on other measures. Predictive Parenting was more expensive than Psychoeducation, with a low probability of being more cost-effective., Conclusion: Feasibility was demonstrated. There was no evidence from this pilot trial that Predictive Parenting resulted in reductions in child EBPs beyond those seen following Psychoeducation; in addition, the effect size was small, and it was more expensive. However, it showed superiority for child compliance and facilitative parenting with moderate effect sizes. Future, definitive studies should evaluate whether augmented or extended intervention would lead to larger improvements., Clinical Trial Registration Information: Autism Spectrum Treatment and Resilience (ASTAR); https://www.isrctn.com/; 91411078., (Copyright © 2021 The Authors. Published by Elsevier Inc. All rights reserved.)
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36. Effect of Preemptive Intervention on Developmental Outcomes Among Infants Showing Early Signs of Autism: A Randomized Clinical Trial of Outcomes to Diagnosis.
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Whitehouse AJO, Varcin KJ, Pillar S, Billingham W, Alvares GA, Barbaro J, Bent CA, Blenkley D, Boutrus M, Chee A, Chetcuti L, Clark A, Davidson E, Dimov S, Dissanayake C, Doyle J, Grant M, Green CC, Harrap M, Iacono T, Matys L, Maybery M, Pope DF, Renton M, Rowbottam C, Sadka N, Segal L, Slonims V, Smith J, Taylor C, Wakeling S, Wan MW, Wray J, Cooper MN, Green J, and Hudry K
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- Early Diagnosis, Female, Humans, Infant, Male, Autism Spectrum Disorder diagnosis, Early Intervention, Educational, Severity of Illness Index
- Abstract
Importance: Intervention for individuals with autism spectrum disorder (ASD) typically commences after diagnosis. No trial of an intervention administered to infants before diagnosis has shown an effect on diagnostic outcomes to date., Objective: To determine the efficacy of a preemptive intervention for ASD beginning during the prodromal period., Design, Setting, and Participants: This 2-site, single rater-blinded randomized clinical trial of a preemptive intervention vs usual care was conducted at 2 Australian research centers (Perth, Melbourne). Community sampling was used to recruit 104 infants aged 9 to 14 months showing early behaviors associated with later ASD, as measured by the Social Attention and Communication Surveillance-Revised. Recruitment occurred from June 9, 2016, to March 30, 2018. Final follow-up data were collected on April 15, 2020., Interventions: Infants were randomized on a 1:1 ratio to receive either a preemptive intervention plus usual care or usual care only over a 5-month period. The preemptive intervention group received a 10-session social communication intervention, iBASIS-Video Interaction to Promote Positive Parenting (iBASIS-VIPP). Usual care comprised services delivered by community clinicians., Main Outcomes and Measures: Infants were assessed at baseline (approximate age, 12 months), treatment end point (approximate age, 18 months), age 2 years, and age 3 years. Primary outcome was the combined blinded measure of ASD behavior severity (the Autism Observation Scale for Infants and the Autism Diagnostic Observation Schedule, second edition) across the 4 assessment points. Secondary outcomes were an independent blinded clinical ASD diagnosis at age 3 years and measures of child development. Analyses were preregistered and comprised 1-tailed tests with an α level of .05., Results: Of 171 infants assessed for eligibility, 104 were randomized; 50 infants (mean [SD] chronological age, 12.40 [1.93] months; 38 boys [76.0%]) received the iBASIS-VIPP preemptive intervention plus usual care (1 infant was excluded after randomization), and 53 infants (mean [SD] age, 12.38 [2.02] months; 32 boys [60.4%]) received usual care only. A total of 89 participants (45 in the iBASIS-VIPP group and 44 in the usual care group) were reassessed at age 3 years. The iBASIS-VIPP intervention led to a reduction in ASD symptom severity (area between curves, -5.53; 95% CI, -∞ to -0.28; P = .04). Reduced odds of ASD classification at age 3 years was found in the iBASIS-VIPP group (3 of 45 participants [6.7%]) vs the usual care group (9 of 44 participants [20.5%]; odds ratio, 0.18; 95% CI, 0-0.68; P = .02). Number needed to treat to reduce ASD classification was 7.2 participants. Improvements in caregiver responsiveness and language outcomes were also observed in the iBASIS-VIPP group., Conclusions and Relevance: Receipt of a preemptive intervention for ASD from age 9 months among a sample of infants showing early signs of ASD led to reduced ASD symptom severity across early childhood and reduced the odds of an ASD diagnosis at age 3 years., Trial Registration: http://anzctr.org.au identifier: ACTRN12616000819426.
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37. Introducing 'Predictive Parenting': A Feasibility Study of a New Group Parenting Intervention Targeting Emotional and Behavioral Difficulties in Children with Autism Spectrum Disorder.
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Hallett V, Mueller J, Breese L, Hollett M, Beresford B, Irvine A, Pickles A, Slonims V, Scott S, Charman T, and Simonoff E
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- Adult, Affective Symptoms diagnosis, Affective Symptoms therapy, Autism Spectrum Disorder diagnosis, Autism Spectrum Disorder therapy, Child, Child Behavior Disorders diagnosis, Child Behavior Disorders therapy, Child, Preschool, Feasibility Studies, Female, Forecasting, Humans, Male, Affective Symptoms psychology, Autism Spectrum Disorder psychology, Child Behavior Disorders psychology, Parenting psychology, Parents psychology
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Parent-mediated interventions can reduce behavioral and emotional problems in children with ASD. This report discusses the development of the first group parent intervention targeting behaviors and anxiety in children with ASD, across the spectrum of cognitive and language ability. 'Predictive Parenting' was developed from the clinical observation (and emerging evidence base) that children with ASD struggle with 'prediction' and anticipating change. It integrates well-established parenting strategies within an ASD-specific framework. The concept was co-created with patient and public involvement panels of parents and adults with ASD. A feasibility study found the programme is acceptable and accessible. Qualitative feedback from participants was largely positive, and critiques were used to inform a larger, pilot randomized controlled trial of the intervention.
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38. Development of the Observation Schedule for Children with Autism-Anxiety, Behaviour and Parenting (OSCA-ABP): A New Measure of Child and Parenting Behavior for Use with Young Autistic Children.
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Palmer M, Paris Perez J, Tarver J, Cawthorne T, Frayne M, Webb S, Baker E, Yorke I, Hay D, Slonims V, Pickles A, Simonoff E, Scott S, and Charman T
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- Anxiety diagnosis, Autistic Disorder diagnosis, Caregivers psychology, Child, Child, Preschool, Emotions, Feasibility Studies, Female, Humans, Male, Parents psychology, Pilot Projects, Anxiety psychology, Autistic Disorder psychology, Behavior Observation Techniques methods, Parenting psychology, Problem Behavior psychology
- Abstract
Co-occurring emotional and behavioral problems (EBPs) frequently exist in young autistic children. There is evidence based on parental report that parenting interventions reduce child EBPs. More objective measures of child EBPs should supplement parent reported outcomes in trials. We describe the development of a new measure of child and parenting behavior, the Observation Schedule for Children with Autism-Anxiety, Behaviour and Parenting (OSCA-ABP). Participants were 83 parents/carers and their 4-8-year-old autistic children. The measure demonstrated good variance and potential sensitivity to change. Child and parenting behavior were reliably coded among verbal and minimally verbal children. Associations between reports from other informants and observed behavior showed the measure had sufficient convergent validity. The measure has promise to contribute to research and clinical practice in autism mental health beyond objective measurement in trials.
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- 2021
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39. The Role of Negative Affectivity in Concurrent Relations Between Caregiver Psychological Distress and Social-Emotional Difficulties in Infants With Early Signs of Autism.
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Chetcuti L, Uljarević M, Varcin KJ, Boutrus M, Wan MW, Slonims V, Green J, Segal L, Iacono T, Dissanayake C, Whitehouse AJO, and Hudry K
- Subjects
- Adult, Cross-Sectional Studies, Emotions, Female, Humans, Infant, Male, Surveys and Questionnaires, Autism Spectrum Disorder diagnosis, Autism Spectrum Disorder psychology, Caregivers psychology, Mothers psychology, Problem Behavior, Psychological Distress, Temperament
- Abstract
Recent evidence suggests the link between caregiver psychological distress and offspring social-emotional difficulties may be accounted for by offspring temperament characteristics. However, existing studies have only focused on neurotypical children; thus, the current study sought to provide an initial examination of this process among children with varying levels of early autism features. Participants included 103 infants aged 9-16 months (M = 12.39, SD = 1.97; 68% male) and their primary caregiver (96% mothers) referred to a larger study by community healthcare professionals. We utilized caregiver-reported measures of psychological distress (Depression Anxiety Stress Scales), infant temperament (Infant Behavior Questionnaire-Revised) and internalizing and externalizing symptoms (Infant-Toddler Social and Emotional Assessment) and administered the Autism Observation Schedule for Infants (AOSI) at an assessment visit to quantify autism features. Infant negative affectivity was found to mediate positive concurrent relations between caregiver psychological distress and infant internalizing and externalizing symptoms, irrespective of the infants' AOSI score. While preliminary and cross-sectional, these results replicate and extend previous findings suggesting that the pathway from caregiver psychological distress to negative affectivity to social-emotional difficulties might also be apparent among infants with varying levels of autism features. More rigorous tests of causal effects await future longitudinal investigation. LAY SUMMARY: Offspring of caregivers experiencing psychological distress (i.e., symptoms of depression, anxiety, and/or stress) may themselves be at increased risk of poor mental health outcomes. Several previous studies conducted with neurotypical children suggest that this link from caregiver-to-child may be facilitated by children's temperament qualities. This study was a preliminary cross-sectional exploration of these relationships in infants with features of autism. We found that infants' elevated negative emotions were involved in the relation between caregiver heightened psychological distress and children's mental health difficulties, consistent with neurotypical development. Autism Res 2020, 13: 1349-1357. © 2020 International Society for Autism Research, Wiley Periodicals, Inc., (© 2020 International Society for Autism Research, Wiley Periodicals, Inc.)
- Published
- 2020
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40. Brief Report: Associations Between Preverbal Social Communication Skills, Language and Symptom Severity in Children with Autism: An Investigation Using the Early Sociocognitive Battery.
- Author
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Taylor LJ, Charman T, Howlin P, Slonims V, and Green J
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- Autistic Disorder psychology, Child, Child, Preschool, Comprehension, Female, Humans, Language, Male, Autistic Disorder diagnosis, Language Development, Neuropsychological Tests standards, Social Skills
- Abstract
We investigated the early sociocognitive battery (ESB), a novel measure of preverbal social communication skills, in children with autism participating in the Paediatric Autism Communication Trial-Generalised (PACT-G). The associations between ESB scores, language and autism symptoms were assessed in 249 children aged 2-11 years. The results show that ESB subscale scores (social responsiveness, joint attention and symbolic comprehension) were significantly associated with concurrent autism symptoms and receptive and expressive language levels. The pattern of association between the ESB subscale scores differed between the ADOS-2 symptom domains and expressive and receptive language. These findings indicate the potential utility of the ESB as a measure of preverbal social communication in children with autism.
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- 2020
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41. Beyond intervention into daily life: A systematic review of generalisation following social communication interventions for young children with autism.
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Carruthers S, Pickles A, Slonims V, Howlin P, and Charman T
- Subjects
- Child, Child, Preschool, Humans, Learning, Male, Autism Spectrum Disorder psychology, Communication, Early Intervention, Educational methods, Generalization, Psychological
- Abstract
Researchers have generally considered autistic individuals to have difficulties generalising learned skills across novel contexts. Successful generalisation is necessary for an intervention to have benefits in everyday life beyond the original learning environment. We conducted a systematic review of randomised controlled trials of early social communication interventions for children with autism in order to explore generalisation and its measurement. We identified nine RCTs that provided evidence of initial target learning and measured generalisation, of which eight demonstrated at least some successful generalisation across people, settings, and/or activities. The findings did not support the widely reported generalisation 'difficulties' associated with autism. However, generalisation was not consistent across all skills within studies, and one study found no generalisation despite evidence for initial target learning within the intervention context. In general, there are few methodologically sound social communication intervention studies exploring generalisation in autism and no consensus on how it should be measured. In particular, failure to demonstrate initial learning of target skills within the intervention setting and an absence of formal mediation analyses of the hypothesised mechanisms limit current research. We outline a framework within which measurement of generalisation can be considered for use in future trials. To maximise the effectiveness of interventions, the field needs to gain a better understanding of the nature of generalisation among autistic individuals and what additional strategies may further enhance learning. Autism Res 2020, 13: 506-522. © 2020 The Authors. Autism Research published by International Society for Autism Research published by Wiley Periodicals, Inc. LAY SUMMARY: It is generally considered that autistic individuals experience difficulties applying things they have learned in one context into different settings (e.g. from school to home). This is important to consider for intervention studies. Our review does not support a complete lack of generalisation but instead suggests that after early social communication intervention, autistic children can transfer some skills to new contexts. Overall, there is limited research in this area and further work is needed., (© 2020 The Authors. Autism Research published by International Society for Autism Research published by Wiley Periodicals, Inc.)
- Published
- 2020
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42. Clinical and cost effectiveness of a parent mediated intervention to reduce challenging behaviour in pre-schoolers with moderate to severe intellectual disability (EPICC-ID) study protocol: a multi-centre, parallel-group randomised controlled trial.
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Farris O, Royston R, Absoud M, Ambler G, Barnes J, Hunter R, Kyriakopoulos M, Oulton K, Paliokosta E, Panca M, Paulauskaite L, Poppe M, Ricciardi F, Sharma A, Slonims V, Summerson U, Sutcliffe A, Thomas M, and Hassiotis A
- Subjects
- Child, Child, Preschool, Cost-Benefit Analysis, England, Humans, Quality of Life, Education, Nonprofessional, Intellectual Disability therapy, Parenting
- Abstract
Background: Children with intellectual disabilities are likely to present with challenging behaviour. Parent mediated interventions have shown utility in influencing child behaviour, although there is a paucity of UK research into challenging behaviour interventions in this population. NICE guidelines favour Stepping Stones Triple P (SSTP) as a challenging behaviour intervention and this trial aims to evaluate its clinical and cost effectiveness in preschool children with moderate to severe intellectual disabilities., Methods: This trial launched in 2017 at four sites across England, with the aim of recruiting 258 participants (aged 30-59 months). The Intervention Group receive nine weeks of SSTP parenting therapy (six group sessions and three individualised face to face or telephone sessions) in addition to Treatment as Usual, whilst the Treatment as Usual only group receive other available services in each location. Both study groups undergo the study measurements at baseline and at four and twelve months. Outcome measures include parent reports and structured observations of behaviour. Service use and health related quality of life data will also be collected to carry out a cost effectiveness and utility evaluation., Discussion: Findings from this study will inform policy regarding interventions for challenging behaviour in young children with moderate to severe intellectual disabilities., Trial Registration Number: Clinicaltrials.gov, NCT03086876. Registered 22nd March 2017, https://clinicaltrials.gov/ct2/show/NCT03086876.
- Published
- 2020
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43. Feasibility study of the National Autistic Society EarlyBird parent support programme.
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Palmer M, San José Cáceres A, Tarver J, Howlin P, Slonims V, Pellicano E, and Charman T
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- Adult, Child, Preschool, Feasibility Studies, Female, Humans, Male, Middle Aged, Societies, Medical, United Kingdom, Young Adult, Autism Spectrum Disorder psychology, Autism Spectrum Disorder therapy, Parents education, Parents psychology, Program Evaluation methods
- Abstract
The EarlyBird programme is a group-based psychoeducation intervention for parents of young children with autism. Although it is widely used in the United Kingdom, the evidence base for the programme is very limited. Using a mixed method, non-randomised research design, we aimed to test (1) the acceptability of the research procedures (recruitment, retention, suitability of measures), (2) the parental acceptability of EarlyBird (attendance, views of the programme, perceived changes) and (3) the facilitator acceptability of EarlyBird (fidelity, views of the programme, perceived changes). Seventeen families with a 2- to 5-year-old autistic child and 10 EarlyBird facilitators took part. Pre- and post-intervention assessment included measures of the child's autism characteristics, cognitive ability, adaptive behaviour, emotional and behavioural problems and parent-reported autism knowledge, parenting competence, stress and wellbeing. Semi-structured interviews were completed at post-intervention with parents and facilitators. For those involved in the study, the research procedures were generally acceptable, retention rates were high and the research protocol was administered as planned. Generally, positive views of the intervention were expressed by parents and facilitators. Although the uncontrolled, within-participant design does not allow us to test for efficacy, change in several outcome measures from pre- to post-intervention was in the expected direction. Difficulties were encountered with recruitment (opt-in to the groups was ~56% and opt-in to the research was 63%), and strategies to enhance recruitment need to be built into any future trial. These findings should be used to inform protocols for pragmatic, controlled trials of EarlyBird and other group-based interventions for parents with young autistic children.
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- 2020
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44. Child and parent outcomes following parent interventions for child emotional and behavioral problems in autism spectrum disorders: A systematic review and meta-analysis.
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Tarver J, Palmer M, Webb S, Scott S, Slonims V, Simonoff E, and Charman T
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- Autism Spectrum Disorder psychology, Child, Child Behavior Disorders psychology, Humans, Problem Behavior psychology, Stress, Psychological etiology, Stress, Psychological psychology, Treatment Outcome, Autism Spectrum Disorder therapy, Behavior Therapy methods, Child Behavior Disorders therapy, Parenting psychology, Parents psychology
- Abstract
There is growing interest in the development of behavioral parent interventions targeting emotional and behavioral problems in children with autism spectrum disorders. Such interventions have potential to improve a number of child and parental well-being outcomes beyond disruptive child behavior. This systematic review and meta-analysis assesses evidence for the efficacy of behavioral parent interventions for disruptive and hyperactive child behavior in autism spectrum disorders, as well as parenting efficacy and stress. A total of 11 articles from nine randomized controlled trials were included. Sufficient data were available to calculate standardized mean difference and show favorable effects of behavioral parent interventions on parent-reported measures of child disruptive behavior (standardized mean difference = 0.67), hyperactivity (standardized mean difference = 0.31) and parent stress (standardized mean difference = 0.37); effects on parent efficacy are less clear (standardized mean difference = 0.39, p = 0.17). There were insufficient data to explore intervention effects on internalizing behavior in autism spectrum disorders, parenting behaviors, or observational and teacher-reported outcomes, providing important avenues for future research. This review adds to growing evidence of the efficacy of behavioral parent interventions for child behavior and parental well-being in autism spectrum disorders (Prospero: CRD42016033979).
- Published
- 2019
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45. Pre-emptive intervention versus treatment as usual for infants showing early behavioural risk signs of autism spectrum disorder: a single-blind, randomised controlled trial.
- Author
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Whitehouse AJO, Varcin KJ, Alvares GA, Barbaro J, Bent C, Boutrus M, Chetcuti L, Cooper MN, Clark A, Davidson E, Dimov S, Dissanayake C, Doyle J, Grant M, Iacono T, Maybery M, Pillar S, Renton M, Rowbottam C, Sadka N, Segal L, Slonims V, Taylor C, Wakeling S, Wan MW, Wray J, Green J, and Hudry K
- Subjects
- Australia, Child Language, Feedback, Female, Humans, Infant, Male, Parents education, Single-Blind Method, Videotape Recording, Autism Spectrum Disorder diagnosis, Autism Spectrum Disorder therapy, Communication, Parent-Child Relations
- Abstract
Background: Great interest exists in the potential efficacy of prediagnostic interventions within the autism spectrum disorder prodrome, but available evidence relates to children at high familial risk. We aimed to test the efficacy of a pre-emptive intervention designed for infants showing early behavioural signs of autism spectrum disorder., Methods: In this single-blind, randomised controlled trial done at two specialist centres in Australia, infants aged 9-14 months were enrolled if they were showing at least three early behavioural signs of autism spectrum disorder on the Social Attention and Communication Surveillance-Revised (SACS-R) 12-month checklist. Infants were randomly assigned (1:1) to receive a parent-mediated video-aided intervention (iBASIS-VIPP) or treatment as usual. Group allocation was done by minimisation, stratified by site, sex, age, and the number of SACS-R risk behaviours. Assessments were done at baseline (before treatment allocation) and at the 6 month endpoint. The primary outcome was Autism Observation Scale for Infants (AOSI), which measures early behavioural signs associated with autism spectrum disorder. Secondary outcomes were a range of infant and caregiver outcomes measured by Manchester Assessment of Caregiver-Infant interaction (MACI), Mullen Scales of Early Learning (MSEL), Vineland Adaptive Behaviour Scales, 2nd edition (VABS-2), MacArthur-Bates Communicative Development Inventory (MCDI), and Parenting Sense of Competence (PSOC) scale. This trial is registered with Australian New Zealand Clinical Trials Registry, number ANZCTR12616000819426., Findings: Between June 9, 2016, and March 30, 2018, 103 infants were randomly assigned, 50 to the iBASIS-VIPP group and 53 to the treatment-as-usual group. After the intervention, we observed no significant differences between groups on early autism spectrum disorder behavioural signs measured by the AOSI (difference estimate -0·74, 95% CI -2·47 to 0·98). We also observed no significant differences on secondary outcomes measuring caregiver non-directiveness (0·16, -0·33 to 0·65), caregiver sensitive responding (0·24, -0·15 to 0·63), and infant attentiveness (-0·19, -0·63 to 0·25) during parent-child interactions (MACI), as well as on researcher-administered measures of receptive (1·30, -0·48 to 3·08) and expressive language (0·54, -0·73 to 1·80), visual reception (0·31, -0·77 to 1·40), and fine motor skills (0·55, -0·32 to 1·41) using the MSEL. Compared with the treatment-as-usual group, the iBASIS-VIPP group had lower infant positive affect (-0·69, -1·27 to -0·10) on the MACI, but higher caregiver-reported receptive (37·17, 95% CI 10·59 to 63·75) and expressive vocabulary count (incidence rate ratio 2·31, 95% CI 1·22 to 4·33) on MCDI, and functional language use (difference estimate 6·43, 95% CI 1·06 to 11·81) on VABS. There were no significant group differences on caregiver-reported measures of MCDI infant gesture use (3·22, -0·60 to 7·04) and VABS social behaviour (3·28, -1·43 to 7·99). We observed no significant differences between groups on self-reported levels of parenting satisfaction (difference estimate 0·21, 95% CI -0·09 to 0·52), interest (-0·23, -0·62 to 0·16) and efficacy (-0·08, -0·38 to 0·22) on PSOC., Interpretation: A pre-emptive intervention for the autism spectrum disorder prodrome had no immediate treatment effect on early autism spectrum disorder symptoms, the quality of parent-child interactions, or researcher-administered measures of developmental skills. However, we found a positive effect on parent-rated infant communication skills. Ongoing follow-up of this infant cohort will assess longer-term developmental effects., Funding: Western Australia Children's Research Fund, Autism Cooperative Research Centre, La Trobe University, and Angela Wright Bennett Foundation., (Copyright © 2019 Elsevier Ltd. All rights reserved.)
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- 2019
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46. A novel group parenting intervention to reduce emotional and behavioural difficulties in young autistic children: protocol for the Autism Spectrum Treatment and Resilience pilot randomised controlled trial.
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Palmer M, Tarver J, Paris Perez J, Cawthorne T, Romeo R, Stringer D, Hallett V, Mueller J, Breese L, Hollett M, Beresford B, Knapp M, Slonims V, Pickles A, Simonoff E, Scott S, and Charman T
- Subjects
- Autism Spectrum Disorder psychology, Autistic Disorder psychology, Child, Child, Preschool, Female, Humans, Male, Parents education, Parents psychology, Pilot Projects, Randomized Controlled Trials as Topic, Autism Spectrum Disorder therapy, Autistic Disorder therapy, Education, Nonprofessional methods
- Abstract
Introduction: The majority of young autistic children display impairing emotional and behavioural difficulties that contribute to family stress. There is some evidence that behavioural parenting interventions are effective for reducing behavioural difficulties in autistic children, with less evidence assessing change in emotional difficulties. Previous trials have tended to use unblinded parent-report measures as primary outcomes and many do not employ an active control, limiting the conclusions that can be drawn., Methods and Analysis: The Autism Spectrum Treatment and Resilience study is a pilot randomised controlled trial (RCT) testing the specific effect of a 12-week group parenting intervention (Predictive Parenting) on primary and secondary outcomes, in comparison to an attention control condition consisting of psychoeducation parent groups. Following a feasibility study to test research procedures and the interventions, the pilot RCT participants include 60 parents of autistic children aged 4-8 years who are randomised to Predictive Parenting versus the attention control. Measures are administered at baseline and post intervention to assess group differences in child and parent outcomes, costs and service use and adverse events. The primary outcome is an objective measure of child behaviours that challenge during interactions with their parent and a researcher. The trial aims to provide data on recruitment, retention, completion of measures and acceptability of the intervention and research protocol, in addition to providing a preliminary indication of potential efficacy and establishing an effect size that could be used to power a larger-scale efficacy trial. We will also provide preliminary estimates of the cost-effectiveness of the interventions., Ethics and Dissemination: Ethical approval was granted from NHS Camden and Kings Cross Research Ethics Committee (ref: 16/LO/1769) along with NHS R&D approval from South London and Maudsley, Guy's and St Thomas', and Croydon Health Services NHS Trusts. The findings will be disseminated through publication in peer-reviewed journals and presentations at conferences., Trial Registration Number: ISRCTN91411078., Competing Interests: Competing interests: AP declares that he receives royalties from WPS for the Social Communication Questionnaire. The views expressed in this publication are those of the authors and not necessarily those of the NHS, the NIHR or the Department of Health., (© Author(s) (or their employer(s)) 2019. Re-use permitted under CC BY. Published by BMJ.)
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- 2019
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47. Evidence-based pathways to intervention for children with language disorders.
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Ebbels SH, McCartney E, Slonims V, Dockrell JE, and Norbury CF
- Subjects
- Age Factors, Child, Humans, Language Development Disorders diagnosis, Language Development Disorders physiopathology, Treatment Outcome, Child Behavior, Child Language, Critical Pathways, Delivery of Health Care, Integrated, Evidence-Based Medicine, Language Development Disorders therapy, Speech-Language Pathology methods
- Abstract
Background: Paediatric speech and language therapist (SLT) roles often involve planning individualized intervention for specific children, working collaboratively with families and education staff, providing advice, training and coaching and raising awareness. A tiered approach to service delivery is currently recommended whereby services become increasingly specialized and individualized for children with greater needs., Aims: To stimulate discussion regarding delivery of SLT services by examining evidence regarding the effectiveness of (1) intervention for children with language disorders at different tiers and (2) SLT roles within these tiers; and to propose an evidence-based model of SLT service delivery and a flowchart to aid clinical decision-making., Methods & Procedures: Meta-analyses and systematic reviews, together with controlled, peer-reviewed group studies where recent systematic reviews were not available, of interventions for children with language disorders are discussed, alongside the differing roles SLTs play in these interventions. Gaps in the evidence base are highlighted., Main Contribution: The service-delivery model presented resembles the tiered model commonly used in education services, but divides individualized (Tier 3) services into Tier 3A: indirect intervention delivered by non-SLTs, and Tier 3B: direct intervention by an SLT. We report evidence for intervention effectiveness, which children might best be served by each tier, the role SLTs could take within each tier and the effectiveness of these roles. Regarding universal interventions provided to all children (Tier 1) and those targeted at children with language weaknesses or vulnerabilities (Tier 2), there is growing evidence that approaches led by education services can be effective when staff are highly trained and well supported. There is currently limited evidence regarding additional benefit of SLT-specific roles at Tiers 1 and 2. With regard to individualized intervention (Tier 3), children with complex or pervasive language disorders can progress following direct individualized intervention (Tier 3B), whereas children with milder or less pervasive difficulties can make progress when intervention is managed by an SLT, but delivered indirectly by others (Tier 3A), provided they are well trained and supported, and closely monitored., Conclusions & Implications: SLTs have a contribution to make at all tiers, but where prioritization for clinical services is a necessity, we need to establish the relative benefits and cost-effectiveness at each tier. Good evidence exists for SLTs delivering direct individualized intervention and we should ensure that this is available to children with pervasive and/or complex language disorders. In cases where service models are being provided which lack evidence, we strongly recommend that SLTs investigate the effectiveness of their approaches., (© 2018 Royal College of Speech and Language Therapists.)
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- 2019
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48. Public health approaches still have room for individualized services: response to commentaries on 'Evidence-based pathways to intervention for children with language disorders'.
- Author
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Norbury CF, McCartney E, Slonims V, Dockrell JE, and Ebbels SH
- Subjects
- Child, Humans, Language Therapy, Language Disorders, Public Health
- Published
- 2019
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49. Paediatric Autism Communication Therapy-Generalised (PACT-G) against treatment as usual for reducing symptom severity in young children with autism spectrum disorder: study protocol for a randomised controlled trial.
- Author
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Green J, Aldred C, Charman T, Le Couteur A, Emsley RA, Grahame V, Howlin P, Humphrey N, Leadbitter K, McConachie H, Parr JR, Pickles A, Slonims V, and Taylor C
- Subjects
- Age Factors, Autism Spectrum Disorder diagnosis, Autism Spectrum Disorder psychology, Child, Child, Preschool, Female, Humans, Interpersonal Relations, Male, Multicenter Studies as Topic, Parent-Child Relations, Randomized Controlled Trials as Topic, Severity of Illness Index, Time Factors, Treatment Outcome, United Kingdom, Autism Spectrum Disorder therapy, Child Behavior, Communication, Early Intervention, Educational methods, Generalization, Psychological
- Abstract
Background: Prior evidence shows that behaviours closely related to the intervention delivered for autism are amenable to change, but it is more difficult to generalise treatment effects beyond the intervention context. We test an early autism intervention designed to promote generalisation of therapy-acquired skills into home and school contexts to improve adaptive function and reduce symptoms. A detailed mechanism study will address the process of such generalisation. Objective 1 will be to test if the PACT-G intervention improves autism symptom outcome in the home and school context of the intervention as well as in the primary outcome research setting. Objective 2 will use the mechanism analysis to test for evidence of acquired skills from intervention generalizing across contexts and producing additive effects on primary outcome., Methods/design: This is a three-site, two-parallel-group, randomised controlled trial of the experimental treatment plus treatment as usual (TAU) versus TAU alone. Children aged 2-11 years (n = 244 (122 intervention/122 TAU; ~ 82/site) meeting criteria for core autism will be eligible. The experimental intervention builds on a clinic-based Pre-school Autism Communication Treatment model (PACT), delivered with the primary caregiver, combined with additional theory- and evidence-based strategies designed to enhance the generalisation of effects into naturalistic home and education contexts. The control intervention will be TAU., Primary Outcome: autism symptom outcome, researcher-assessed using a standardised protocol., Secondary Outcomes: autism symptoms, child interaction with parent or teacher, language and reported functional outcomes in home and school settings. Outcomes measured at baseline and 12-month endpoint in all settings with interim interaction measurements (7 months) to test treatment effect mechanisms. Primary analysis will estimate between-group difference in primary outcome using analysis of covariance with test of homogeneity of effect across age group. Mechanism analysis will use regression models to test for mediation on primary outcome by parent-child and teaching staff-child social interaction., Discussion: This is an efficacy and mechanism trial of generalising evidence-based autism treatment into home and school settings. It will provide data on whether extending treatment across naturalistic contexts enhances overall effect and data on the mechanism in autism development of the generalisation of acquired developmental skills across contexts., Trial Registration: ISRCTN, ID: 25378536 . Prospectively registered on 9 March 2016.
- Published
- 2018
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50. The Association Between Child and Family Characteristics and the Mental Health and Wellbeing of Caregivers of Children with Autism in Mid-Childhood.
- Author
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Salomone E, Leadbitter K, Aldred C, Barrett B, Byford S, Charman T, Howlin P, Green J, Le Couteur A, McConachie H, Parr JR, Pickles A, and Slonims V
- Subjects
- Adolescent, Adult, Autistic Disorder epidemiology, Child, Child, Preschool, Emotions, Female, Humans, Intellectual Disability, Male, Autistic Disorder psychology, Caregivers psychology, Family Characteristics, Mental Health
- Abstract
We examined predictors of mental health difficulties and wellbeing in caregivers of children with autism in the Pre-school Autism Communication Trial cohort in middle childhood (N = 104). Child's intellectual disability, daily living skills impairment, elevated emotional and behavioural difficulties, high educational level of caregiver and household income below the median significantly predicted caregivers' mental health difficulties, but autism severity, child communication skills and family circumstances did not. Lower caregiver mental wellbeing was predicted by elevated child emotional and behavioural difficulties. The need to support the mental health and wellbeing of caregivers of children with autism is discussed in light of the results.
- Published
- 2018
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