Catherine Aldred

Parent-mediated communication-focused treatment in children with autism (PACT): a randomised controlled trial

Summary Background Results of small trials suggest that early interventions for social communication are effective for the treatment of autism in children. We therefore investigated the efficacy of such an intervention in a larger trial. Methods Children with core autism (aged 2 years to 4 years and 11 months) were randomly assigned in a one-to-one ratio to a parent-mediated communication-focused (Preschool Autism Communication Trial [PACT]) intervention or treatment as usual at three specialist centres in the UK. Those assigned to PACT were also given treatment as usual. Randomisation was by use of minimisation of probability in the marginal distribution of treatment centre, age (≤42 months or >42 months), and autism severity (Autism Diagnostic Observation Schedule-Generic [ADOS-G] algorithm score 12–17 or 18–24). Primary outcome was severity of autism symptoms (a total score of social communication algorithm items from ADOS-G, higher score indicating greater severity) at 13 months. Complementary secondary outcomes were measures of parent-child interaction, child language, and adaptive functioning in school. Analysis was by intention to treat. This study is registered as an International Standard Randomised Controlled Trial, number ISRCTN58133827. Results 152 children were recruited. 77 were assigned to PACT (London [n=26], Manchester [n=26], and Newcastle [n=25]); and 75 to treatment as usual (London [n=26], Manchester [n=26], and Newcastle [n=23]). At the 13-month endpoint, the severity of symptoms was reduced by 3·9 points (SD 4·7) on the ADOS-G algorithm in the group assigned to PACT, and 2·9 (3·9) in the group assigned to treatment as usual, representing a between-group effect size of −0·24 (95% CI −0·59 to 0·11), after adjustment for centre, sex, socioeconomic status, age, and verbal and non-verbal abilities. Treatment effect was positive for parental synchronous response to child (1·22, 0·85 to 1·59), child initiations with parent (0·41, 0·08 to 0·74), and for parent-child shared attention (0·33, −0·02 to 0·68). Effects on directly assessed language and adaptive functioning in school were small. Interpretation On the basis of our findings, we cannot recommend the addition of the PACT intervention to treatment as usual for the reduction of autism symptoms; however, a clear benefit was noted for parent-child dyadic social communication. Funding UK Medical Research Council, and UK Department for Children, Schools and Families.

ArticlesParent-mediated communication-focused treatment in children with autism (PACT): a randomised controlled trial

Summary Background Results of small trials suggest that early interventions for social communication are effective for the treatment of autism in children. We therefore investigated the efficacy of such an intervention in a larger trial. Methods Children with core autism (aged 2 years to 4 years and 11 months) were randomly assigned in a one-to-one ratio to a parent-mediated communication-focused (Preschool Autism Communication Trial [PACT]) intervention or treatment as usual at three specialist centres in the UK. Those assigned to PACT were also given treatment as usual. Randomisation was by use of minimisation of probability in the marginal distribution of treatment centre, age (≤42 months or >42 months), and autism severity (Autism Diagnostic Observation Schedule-Generic [ADOS-G] algorithm score 12–17 or 18–24). Primary outcome was severity of autism symptoms (a total score of social communication algorithm items from ADOS-G, higher score indicating greater severity) at 13 months. Complementary secondary outcomes were measures of parent-child interaction, child language, and adaptive functioning in school. Analysis was by intention to treat. This study is registered as an International Standard Randomised Controlled Trial, number ISRCTN58133827. Results 152 children were recruited. 77 were assigned to PACT (London [n=26], Manchester [n=26], and Newcastle [n=25]); and 75 to treatment as usual (London [n=26], Manchester [n=26], and Newcastle [n=23]). At the 13-month endpoint, the severity of symptoms was reduced by 3·9 points (SD 4·7) on the ADOS-G algorithm in the group assigned to PACT, and 2·9 (3·9) in the group assigned to treatment as usual, representing a between-group effect size of −0·24 (95% CI −0·59 to 0·11), after adjustment for centre, sex, socioeconomic status, age, and verbal and non-verbal abilities. Treatment effect was positive for parental synchronous response to child (1·22, 0·85 to 1·59), child initiations with parent (0·41, 0·08 to 0·74), and for parent-child shared attention (0·33, −0·02 to 0·68). Effects on directly assessed language and adaptive functioning in school were small. Interpretation On the basis of our findings, we cannot recommend the addition of the PACT intervention to treatment as usual for the reduction of autism symptoms; however, a clear benefit was noted for parent-child dyadic social communication. Funding UK Medical Research Council, and UK Department for Children, Schools and Families.

Preschoolers with autism show greater impairment in receptive compared with expressive language abilities

BACKGROUND In early typical language development, children understand words before they are able to use them in speech. Children with autism spectrum disorders (ASD) generally show impairments in both the comprehension and the production of language. However, the relative degree of delay or impairment in each of these sub-domains may also be atypical and remains less well-understood. AIMS Relative delay in receptive and expressive language skills was examined within a large sample of preschoolers with autism. Childrens language abilities varied from pre-verbal to fluent speech. METHOD & PROCEDURES Scores on one direct clinician assessment and two parent-report measures of language were obtained for 152 preschoolers with core autism. OUTCOMES & RESULTS As expected, on average, the language ability of the children with autism was lower than typical age norms, albeit with substantial individual variability. On all three language measures, receptive ability was relatively more impaired than expressive ability. Higher non-verbal ability was associated with such an atypical language profile. CONCLUSIONS & IMPLICATIONS Recognition of the marked receptive language impairment relative to expressive language, found to affect at least one-third of preschoolers with autism in this sample, has important implications for interacting with these children and for informing appropriate targets in language and communication intervention.

Parent-mediated social communication therapy for young children with autism (PACT): long-term follow-up of a randomised controlled trial

Summary 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 Cohens 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.

Treatment mechanism in the MRC preschool autism communication trial: implications for study design and parent-focussed therapy for children.

BACKGROUND The PACT randomised-controlled trial evaluated a parent-mediated communication-focused treatment for children with autism, intended to reduce symptom severity as measured by a modified Autism Diagnostic Observation Schedule-Generic (ADOS-G) algorithm score. The therapy targeted parental behaviour, with no direct interaction between therapist and child. While nonsignificant group differences were found on ADOS-G score, significant group differences were found for both parent and child intermediate outcomes. This study aimed to better understand the mechanism by which the PACT treatment influenced changes in child behaviour though the targeted parent behaviour. METHODS Mediation analysis was used to assess the direct and indirect effects of treatment via parent behaviour on child behaviour and via child behaviour on ADOS-G score. Alternative mediation was explored to study whether the treatment effect acted as hypothesised or via another plausible pathway. Mediation models typically assume no unobserved confounding between mediator and outcome and no measurement error in the mediator. We show how to better exploit the information often available within a trial to begin to address these issues, examining scope for instrumental variable and measurement error models. RESULTS Estimates of mediation changed substantially when account was taken of the confounder effects of the baseline value of the mediator and of measurement error. Our best estimates that accounted for both suggested that the treatment effect on the ADOS-G score was very substantially mediated by parent synchrony and child initiations. CONCLUSIONS The results highlighted the value of repeated measurement of mediators during trials. The theoretical model underlying the PACT treatment was supported. However, the substantial fall-off in treatment effect highlighted both the need for additional data and for additional target behaviours for therapy.

Exploring the effects of communication intervention for developmental pragmatic language impairments: a signal‐generation study

BACKGROUND The remediation of pragmatic problems forms a significant part of the caseload for professionals working with children with communication problems. There is little systematic evidence that demonstrates the benefits of speech and language therapy for children whose difficulties lie primarily within the pragmatic domain or which indicates whether changes in pragmatic behaviours, which are a result of a specific intervention, can be measured over time. AIMS To generate a signal of change in pragmatic and other language behaviours for children with pragmatic language impairments; to gauge the magnitude and nature of the signal and to make recommendations for future studies. METHODS AND PROCEDURES A case series of six children with pragmatic language impairments without diagnosis of autism received 8 weeks of individual intensive speech and language therapy supported in a mainstream educational setting in the UK. Measures of pragmatic behaviours in conversation were made at seven data points before and after therapy using Bishops ALICC procedure. Conversation coders were blind to the point of assessment. Inferential comprehension, narrative, sentence formulation and sentence recall skills were also tested before and after therapy. The opinions of teachers and parents were sought regarding any change in communication and social abilities of the children over time. OUTCOMES AND RESULTS All children showed change in communication behaviour on some conversational measures, even if the child functioned at the ceiling on standardized language testing. Some conversation measures had more utility as outcome measures than others. Most children showed substantial change on standardized language measures, but there are limitations on the use of these due to heterogeneity within the group. Overall, the intervention produced a signal for change in pragmatics and/or language behaviour in all children. Parent/teacher opinion reported demonstrable change in communication behaviour and engagement in the curriculum. CONCLUSIONS There is a strong signal that change in pragmatic language behaviour can be measured in well-controlled intervention studies but this signal is complex. Outcome measures should take into account changes in language processing skills that are significantly impaired in many children with PLI. For those children within the PLI group who function at ceiling on language tests, conversational measures may have the potential to signal change, but this finding has not been subjected to group study or to testing in generalized settings. Qualitative data regarding behaviour, classroom engagement and generalization of language gains will be an essential supplement to measuring progress in a diverse population.

Effectiveness of the parent-mediated intervention for children with autism spectrum disorder in south Asia in India and Pakistan (PASS): a randomised controlled trial

BACKGROUND Autism spectrum disorder affects more than 5 million children in south Asia. Although early interventions have been used for the treatment of children in high-income countries, no substantive trials have been done of the interventions adapted for use in low-income and middle-income countries (LMICs). We therefore assessed the feasibility and acceptability of the parent-mediated intervention for autism spectrum disorder in south Asia (PASS) in India and Pakistan. METHODS A single-blind randomised trial of the comparison of 12 sessions of PASS (plus treatment as usual) with treatment as usual alone delivered by non-specialist health workers was done at two centres in Goa, India, and Rawalpindi, Pakistan. Children aged 2-9 years with autism spectrum disorder were randomly assigned (1:1) by use of probabilistic minimisation, controlling for treatment centre (Goa or Rawalpindi), age (<6 years or ≥6 years), and functional impairment (Vineland Adaptive Behaviour Scale Composite score <65 or ≥65). The primary outcome was quality of parent-child interaction on the Dyadic Communication Measure for Autism at 8 months. Analysis was by intention to treat. The study is registered with ISRCTN, number ISRCTN79675498. FINDINGS From Jan 1 to July 30, 2013, 65 children were randomly allocated, 32 to the PASS group (15 in Goa and 17 in Rawalpindi) and 33 to the treatment-as-usual group (15 in Goa and 18 in Rawalpindi). 26 (81%) of 32 participants completed the intervention. After adjustment for minimisation factors and baseline outcome, the primary outcome showed a treatment effect in favour of PASS in parental synchrony (adjusted mean difference 0·25 [95% CI 0·14 to 0·36]; effect size 1·61 [95% CI 0·90 to 2·32]) and initiation of communication by the child with the parent (0·15 [0·04 to 0·26]; effect size 0·99 [0·29 to 1·68]), but time in mutual shared attention was reduced (-0·16 [-0·26 to -0·05]; effect size -0·70 [-1·16 to -0·23]). INTERPRETATION Our results show the feasibility of adapting and task-shifting an intervention used in a high-income context to LMICs. The findings also replicate the positive primary outcome treatment effects of a parent-mediated communication-focused intervention in the original UK Preschool Autism Communication Trial, with one negative effect not reported previously. FUNDING Autism Speaks, USA.

Pragmatic language impairment: case studies of social and pragmatic language therapy

The current position on speech and language intervention for children who have pragmatic language impairment (PLI) is limited by a lack of evidence to support practice. Two intervention outcome case studies of children with PLI, aimed at establishing efficacy, are presented in this paper Standardized language tests and conversational sampling were used to assess the children pre-and post-therapy. Each child received eight weeks of intervention, three times a week, from a specialist speech and language therapist. This experi-mental treatment, which was funded as part of a research project, targeted social adaptation skills of the child and adults in his communication environment, in addition to work on communication acts, conversation and narrative skills and facilitating understanding of social inference. One child, with isolated social andpragmatic difficulties, showed measured and reported improvements in conversational skills. The second child, who has additional language disorder, showed changes in language processing skills but no changes in pragmatic abilities. The implications for choosing interventions, for the training of practitioners and questions for further research are discussed. The therapy resources used in intervention are listed.

Adapting an evidence-based intervention for autism spectrum disorder for scaling up in resource-constrained settings: the development of the PASS intervention in South Asia

Background Evidence-based interventions for autism spectrum disorders evaluated in high-income countries typically require highly specialised manpower, which is a scarce resource in most low- and middle-income settings. This resource limitation results in most children not having access to evidence-based interventions. Objective This paper reports on the systematic adaptation of an evidence-based intervention, the Preschool Autism Communication Therapy (PACT) evaluated in a large trial in the United Kingdom for delivery in a low-resource setting through the process of task-shifting. Design The adaptation process used the Medical Research Council framework for the development and adaptation of complex interventions, focusing on qualitative methods and case series and was conducted simultaneously in India and Pakistan. Results The original intervention delivered by speech and language therapists in a high-resource setting required adaptation in some aspects of its content and delivery to enhance contextual acceptability and to enable the intervention to be delivered by non-specialists. Conclusions The resulting intervention, the Parent-mediated intervention for Autism Spectrum Disorder in South Asia (PASS), shares the core theoretical foundations of the original PACT but is adapted in several respects to enhance its acceptability, feasibility, and scalability in low-resource settings.

Parents’ perspectives on care of children with autistic spectrum disorder in South Asia – Views from Pakistan and India

Abstract Autism spectrum disorder (ASD) affects about 1.4% of the population in South Asia but very few have access to any form of health care service. The objective of this study was to explore the beliefs and practices related to the care of children with ASD to inform strategies for intervention. In Pakistan, primary data were collected through in-depth interviews of parents (N = 15), while in India a narrative review of existing studies was conducted. The results show that the burden of care is almost entirely on the mother, leading to high levels of stress. Poor awareness of the condition in both family members and front-line health-providers leads to delay in recognition and appropriate management. There is considerable stigma and discrimination affecting children with autism and their families. Specialist services are rare, concentrated in urban areas, and inaccessible to the majority. Strategies for intervention should include building community and family support networks to provide respite to the main carer. In the absence of specialists, community members such as community health workers, traditional practitioners and even motivated family members could be trained in recognizing and providing evidence-based interventions. Such task-shifting strategies should be accompanied by campaigns to raise awareness so greater inclusivity can be achieved.